JBR 2014-4 by office

WETTEREN 1

P 702083

Volume 97 Page 197-270 July-August

Bimonthly

–

2014

DIAGNOSTIC AND INTERVENTIONAL IMAGING, RELATED IMAGING SCIENCES, AND CONTINUING EDUCATION

ORGANE DE LA SOCIETE BELGE DE RADIOLOGIE (SBR) ORGAAN VAN DE BELGISCHE VERENIGING VOOR RADIOLOGIE (BVR) 00a-Couv-2014 (4).indd 1

14/08/14 14:14

Subscribers’ information The JBR-BTR is published 6 times a year. Subscription of members of the Belgian Society of Radiology are included in membership dues and are handled by the Society. Non-members’ subscriptions are available from the ARSMB-KVBMG. The rate is valid to date and can be amended without notice according to fluctuation of printing and material costs. Annual subscriptions or single issue orders should be made promptly. The publishers cannot guarantee supply of back issues. Change of address must be notified 60 days in advance. RATES: Annual Single issue Belgium 150 € 38 € Other Countries 175 € 44 € All amounts are net and include postal and handling charges. You are kindly invited to address all your correspondence to Mrs A. Hirsch and execute all payments to ARSMBKVBMG (see below).

Instructions aux abonnés Le JBR-BTR publie 6 fascicules par an. Les tarifs sont susceptibles de modifications sans préavis, en fonction de l’évolution des prix du marché du papier et des travaux d’impression. Le prix de l’abonnement des membres de la Société Royale de Radiologie est inclus dans le montant de la cotisation. L’abonnement d’un non-membre est à souscrire auprès de l’ARSMB. La souscription d’abonnement ou la commande de numéro isolé doit être exécutée rapidement, l’éditeur ne pouvant pas garantir la livraison d’éditions passées. Les changements d’adresse doivent être signalés 60 jours à l’avance. TARIF: Annuel Fascicule Belgique 175 € 42 € Autres pays 200 € 49 € Envoi et port inclus. Nous vous prions d’adresser toute correspondance à Mme A. Hirsch et d’effectuer tout paiement au compte de l’ARSMB-KVBMG (voir ci-dessous).

Instructies voor abonnees Het JBR-BTR geeft 6 nummers uit per jaar. Het tarief is vatbaar voor wijzigingen zonder voorafgaand bericht, in verhouding tot de evolutie van de papierprijzen en loonkosten in de grafische nijverheid. Het abonnement van de leden van de Koninklijke Vereniging voor Radiologie is begrepen in de bijdrage van het lidgeld. De abonnementen van niet-leden zijn te onderschrijven bij de KVBMG. Jaarabonnementen of bestellingen van losse nummers moet zo snel mogelijk gebeuren, de uitgever waarborgt de levering van de vorige nummers niet voor de abonnementen die te laat werden onderschreven. De adresveranderingen moeten 60 dagen te voren gemeld worden. TARIEF: Jaarlijks Aflevering Belgie 175 € 42 € Andere landen 200 € 49 € Verzendingskosten zijn inbegrepen. U wordt vriendelijk verzocht alle briefwisseling te richten aan Mevr. A. Hirsch en alle betalingen te verrichten op het banknummer van ARSMB-KVBMG (zie hieronder).

Association Royale des Sociétés Scientifiques Médicales Belges – (ARSMB), asbl avenue W. Churchill 11/30, B-1180 Bruxelles, Belgique tél.: (02) 374 25 55 fax: (02) 374 96 28

02-voorblz-2014-1.indd 1

Koninklijke Vereniging van de Belgische Medische Wetenschappelijke Genootschappen – (KVBMG), vzw W. Churchill-laan 11/30, B-1180 Brussel, België tel.: (02) 374 25 55 fax: (02) 374 96 28

Webaddress: http://www.ulb.ac.be/medecine/loce/amb.htm E-mail: jbr-btr@skynet.be Bank Account: Post Office Account Fortis: 210-0251210-32 Giro: 000-0273502-59 IBAN: BE 90210025121032 IBAN: BE 84000027350259 BIC: GE BABEBB36A BIC: BPOTBEB1

5/08/14 15:57

JBR-BTR  97/4  2014 Journal Belge de  Belgisch Tijdschrift voor  RADIOLOGIE

Founded in 1907 A bimonthly journal devoted to diagnostic and interventional imaging, related imaging sciences, and continuing education Contents Percutaneous cholecystostomy: single centre experience in 111 patients with an acute cholecystitis R. Peters, B. Peters, M. Simoens, S. Braak. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 197 How sensitive and specific are MRI features of sacroiliitis for diagnosis of spondyloarthritis L. Jans, L. Coeman, L. Van Praet, P. Carron, D. Elewaut, F. Van den Bosch, J.L. Jaremko, W. Huysse, K.L. Verstraete. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 202 Real time US guided pediatric percutaneous renal biopsy: the traditional method vs angled tangential approach K. Can Caliskan, G. Ozcelik, E. Cakmakci, S. Mert Ulussay, A. Senol Celebi, S. Turk, A. Ozagari, Z. Karpat . . . . 206 Diffusion-weighted MRI: role in the differential diagnosis of breast lesions C. Altay, P. Balci, S. Altay, S. Karasu, S. Saydam, T. Canda, O. Dicle. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 211 Mucocele of the appendix : a case report and review of the literature M. Faure, R. Salgado, B. Op de Beeck, J.L. Termote, P.M. Parizel. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 217 Update on MR imaging of spondyloarthritis. Part one: The sacro-iliac joint B.C. Vande Berg, P. Omoumi, A. Larbi, F. Lecouvet, J. Malghem. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 222 Update on MR imaging of spondyloarthritis. Part two: spine MR imaging B.C. Vande Berg, P. Omoumi, A. Larbi, F. Lecouvet, J. Malghem. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 228 Capillary telangiectasia of the brain: imaging with various MR techniques F. Gelal, L. Karakas, A. Sarsilmaz, K. Yucel, C. Dundar, M. Apaydin. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 233 Small bowel angioedema induced by angiotensin converting enzyme (ACE) inhibitor: US and CT findings M.L. Coelho, R. Amaral, L. Curvo-Semedo, F. Caseiro-Alves. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 239 Added value of diffusion weighted MRI in the diagnosis of postpartum ovarian vein thrombosis K. De Cuyper, M. Eyselbergs, P. Bernard, L. Clabout, F.M. Vanhoenacker. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 242 Angioleiomyoma in the soleus muscle A. Van Holsbeeck, L. Van den Daelen, E. Steenkiste, B. Van Holsbeeck . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 245 Primary undifferentiated embryonal sarcoma of the liver misdiagnosed as hydatid cyst in a child: case report and review of the literature A.M. Halefoglu, A. Oz. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 248 Appearance and vanishing of a spinal intradural arachnid cyst after multiple epidural corticosteroid injections. A spontaneously resolving cause of symptomatic lumbar stenosis P. Mailleux, G. Milbouw, G. Koerts, P. Verbeeck, X. Willems. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 251 Hepatic angiosarcoma occurring 65 years after thorium dioxide (Thorostrast) exposure: imaging, surgical and histopathologic findings of a historical case B. Coulier, F. Pierard, I. Gielen, Ph. Maldague. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 254 Fibrous pseudotumor of the tunica vaginalis: contrast enhanced sonography with pathologic correlation Th. Puttemans, M.I. Ingabire, J.L. Jorion, A.Ph. Draguet . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 259

IMAGES IN CLINICAL RADIOLOGY An unusual presentation of a pelvic textiloma mimicking a tumor R. Kadi, V. Massard, K. Vanden Houte, C. Gerard, L. Divano, J. Jani, M. Laureys, E. Najar, M. Cannie. . . . . . . . 262 Leiomyoma of the urinary bladder T. Dewaele, L. D’Hooghe, S. Weters, P. Devisschere . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 263 Imaging findings in a patient with saliva in the auditory canal P. Gillardin, P. Tomassen, Ch. Vanclooster, P. Vander Eecken, M. Lemmerling. . . . . . . . . . . . . . . . . . . . . . . . . . . 264 MRI features of acute bilateral caudate infarcts in pregnant woman M. Semnic, R. Semnic, K. Petrovic, F.M. Vanhoenacker. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 265

01-JBR-contents-14-4.indd 1

14/08/14 14:13

Pseudo-achalasia: a complication of laparoscopic adjustable gastric banding P. Terryn, J. Pringot, L. Ghijselings, B. Bormans, P. Matthys. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 266 A case of acute appendicitis at atypical localization I. Basara, M. Secil. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 267 Spontaneous osteonecrosis of the knee (SONK) A.M. Filip, S.B. Van den Broeck. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 268

LETTER TO THE EDITOR US diagnosis of acute pancreatitis caused by ruptured hydatid disease to the biliary system B. Karaman, B. Battal, S. Sari, S. Verim. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 269 Abstract of papers for full membership at the Belgian Society of Radiology Conebeam-CT and fluoroscopy guided percutaneous absolute alcohol sclerotherapy of aneurysmal bone cyst â&#x20AC;&#x201C; A single centre experience J. De Groote, T. Dewaele, L. Defreyne . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 270 News from the Vascular and Interventional Imaging section . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 271 Annual BSR Symposium: Programme . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . v Forthcoming Courses and Meetings. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . vi Instructions to Authors. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . iii Subscribers information. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . cii Advertising index . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 270

u The terms used for indexation of subjects were developed by the Radiological Society of North America (RSNA) over a period of years. Their use here is by permission of the RSNA. The terms may not be used in any other index, print or electronic, except by specific permission of RSNA. uu Indexed in Index Medicus and in Zentralblatt Radiologie. Evaluated for Medline User, EMBASE and CANCERNET. Abstracted in Excerpta Medica Journals.

01-JBR-contents-14-4.indd 2

14/08/14 14:13

Editor-in-Chief: J. Pringot

Board of the Belgian Society of Radiology:

Consulting Co-Editor: Ph. Grenier (Paris)

President: G. Villeirs

Managing Editors: P. Seynaeve

Vice-President: D. Henroteaux

Editorial Board: F. Avni, L. Breysem, N. Buls, B. Coulier, B. Daenen, E. Danse, H. Degryse, P. Demaerel, B. Ghaye, J. Gielen, P. Habibollahi, N. Hottat, M. Laureys, F. Lecouvet, M. Lemmerling, B. Lubicz, J.F. Monville, T. Mulkens, A. Nchimi, J.F. Nisolle, B. Op de Beeck, R. Oyen, S. Pans, V.P. Parashar (USA), P. Parizel, P. Peene, H. Rigauts, N. Sadeghi, P. Simoni, S. Sintzoff Jr, A. Snoeckx, J. Struyven, H. Thierens, P. Van Dyck, F. Vanhoenacker, Ph. Van Hover, J. Verschakelen, K. Verstraete.

Secretary: Ch. Delcour Treasurer: P. Vanhoenacker BVAS/ABSYM-Representative: O. Ghekiere Board Members: P. Aerts, B. De Foer, J.-F. De Wispelaere, M. Grieten, H. Jaspers, J.-P. Joris, R. Oyen, P. Seynaeve, G. Souverijns, D. Tack, B. Vandeberg, G. Vandenbosch, Ch. Van de Velde

Scientific Committee Members: R. Achten, D. Bielen, B. De Foer, Y. Lefebvre, M. Lemort, J. Pringot, R. Salgado, D. Tack, J. Ver schakelen

President: R. Oyen Secretaries: J. de Mey, B. Vande Berg

Sections of the Belgian Radiological Society (BSR): Abdominal and digestive imaging Bone and joints Breast imaging Cardiac imaging Cardiovascular and interventional radiology Chest radiology Head and neck radiology Neuroradiology Pediatric radiology

B. Op de Beeck, E. Danse J.F. Nisolle, M. Shahabpour M. Mortier, S. Murgo N. Mollet, A. Nchimi S. Heye, D. Henroteaux B. Ghaye, W. De Wever J. Widelec, R. Hermans M. Lemmerling, L. Tshibanda B. Desprechins, L. Breysem

For addresses and particulars, see website at http://www.rbrs.org Instructions to authors The purpose of The Belgian Journal of Radio logy is the publication of articles dealing with diagnostic radiology and related imag ing techniques, therapeutic radiology, allied sciencesand continuing education. All — new and revised — manuscripts and correspond ence should be addressed to JBR-BTR Edito rialOffice, Avenue W. Churchill 11/30, B-1180 Bruxelles, tel.: 02-374 25 55, fax: 32-2-374 96 28. Please note that the following instructions are based on the “Uniform Requirements for manuscripts Submitted to Biomedical Jour nals” adopted by the International Committee of Medical Journal Editors (Radiology, 1980,135: 239-243). It should however be noted that presentation modifications may be introduced by the Editorial Office in order to conform with the JBR-BTR personal style. Authors should specify to which of the fol lowing headings their manuscript is intended: Original Article, Review Article, Case Report, Pictorial Essay, Continuing Education, Technical Note, Book Review, Opinion, Letter to the Editor, Comment, Meeting News, in Memoriam, News. Authors should consider the following remarks and submit their manuscripts accord ingly. All articles must contain substantive and specific scientific material. – Original articles are articles dealing with one specific area of Radiology or allied sciencerelated through the personal expe rience of the author. – Review articles are special articles reporting the experience of the author considered in

02-voorblz-2014-1.indd 2

the general perspective of the literature over the topic. – Case reports are short descriptions of a particular case providing a message directly linked to an individual patient investigated. – No more than one case should be described in detail and clinical description should be kept to a minimum. Case reports should invest the usual headings of articles but should focus on the particular radiologic procedure that contributed to the diagno sis. References should be present, though limited in number. Tables and acknowl edgements are usually omitted. – Pictorial essays are articles presenting information through illustrations and leg ends. The presentation remarks stated in the paragraph dealing with case reports apply to pictorial essays. – Continuing education articles are designed in accordance with the general guidelines for articles published in the JBR-BTR in particular they are divided into introduc tion, material and methods, results, discus sion, references, and are provided with an abstract. However, papers addressing the continuing education may have only additionnally to their contents an introduction (stating the aim of the article and providing any back ground information useful to understand why the topic is relevant, and describing the subtopics covered by the study), refer ences, and an abstract. Tables should be limited to a maximum of one table per 6 pages of manuscript. Illustrations should also be limited to a maximum of one illustration (1010 cm)

(possibly made up of different parts) per 3 pages of manuscript. All the material should be made available to the JBR- BTR editorial office (2 copies of the manuscript with 2 sets of illustrations) with the corresponding diskette though there will not be peer review. – Images in Clinical Radiology are short (max. 1 typed page) case reports designed to illustrate with max. 3 figures a specific enti ty. The report should not include abstract nor discussion but consist of a synthetic description of the clinical and radiological features as well as the final diagnosis and one major reference. Technical notes are short descriptions of a specific technique, procedure or equipment of interest to radi ologists. Technical notes may originate from radiologists having experience of the item presented or from commercial firms (these should contact the Editorial Office to obtain specific guidelines for publication). The manuscript length should be inferior to 1 typed page, original language should be English, the manuscript may be accompa nied by maximum 1 b/w figure, and include one major reference. – Book reviews should be limited to one typed page, mention full references of the book, including number of pages, of illus trations (when available), and price. The author should specify to whom the book is intended and give a personal apprecia tion. They will be published with the initial lettersof the signature. (continued on JBR-BTR 2014, 97-2, p. VIII)

14/08/14 13:57

DUAL SOFT BAG INJECTION SYSTEM

This medical device is intended for use by medical imaging and diagnostic health professionals • FlowSens® • Class: II b • CE Certificate: in compliance with directive 93/42/ EEC • Certifying organization: LNE G-Med (CE 0459) • Technical data: CT dual-head soft bag injector - Hydraulic technology - Use with prefilled bags, bags and vials - Installation: ceiling-mount / pedestal - Flow rate: 0.3 to 10mL/s - Maximum pressure: 21bar / 2100kPa / 305PSI - Limit of pressure: programmable - Pressure monitoring: graphical & numerical Bag maximum capacity: 500mL - Number of phases: 6 - Volume per phase: 1 to 220mL - Air detection: automatic - Number of filling speeds: 3 - Filling: manual / automatic - Priming: manual / automatic - Delay between phases: 0 to 900s - Protocol programming modes: 3 - Memory capacity (protocols): 20 libraries, 4000 protocols - Memory capacity (historical & statistics): 24000 injections, 300000 events - Voltage: 230V AC - Power frequency: 50-60Hz - Power consumption: 1000VA - Injector pedestal dimensions / weight (H x W x D): 1422 x 750 x 750mm / 75kg - Injector ceiling mount dimensions / weight (H x W x D): 1016 x 358 x 503mm / 60kg - Remote console dimensions / weight (H x W x D): 355 x 300 x 249mm / 3,5kg - Power unit dimensions / weight (H x W x D): 654 x 668 x 356mm / 70kg - Complying with European Directive 2002/96/EC, amended by Directive 2012/96/EC (WEEE) • Manufacturer: MEDEX - 240 allée Jacques Monod - 69800 Saint-Priest - France • Document creation date: 2014 • For complete information about precautions and optimal usage conditions for this medical device, we recommend consulting the instructions notice/user’s manual.

Flowsens_Teasing1-2 A4-2014OK.indd 2

Conception : ANNAPURNA 8000 - Photos : Guerbet®, Shutterstock.

A high flying simplicity

02/12/13 16:18

Annual symposium

RQ IHH PHPEHUV %65 EHUV sts in training

Belgian Society of Radiology (BSR)

November 15th, 2014

LWV LQFOXGLQJ HWKLFV HFRQRP\

Meeting venue

ospital nstraat 1 - 1120 Brussels >0LOLWDU\ +RVSLWDO@ IROORZ WUDIÂżF VLJQV

Meeting venue : : Meeting venue

K K Welcome K K Recist and beyond ( &RFKH 8&/ %UX[HOOHV K K MRI and ovarian cancer: what to know, what to do? 9 9DQGHFDYH\H .8 /HXYHQ K K Assessment of tumor response to treatment using PET/CT 5 +XVWLQ[ 8OJ 6DUW 7LOPDQ K K Hepatocellular carcinoma endovascular treatment: what the radiologist needs to know - *RO]DULDQ 0LQQHDSROLV 8QLYHUVLW\ K K Image fusion during vascular and nonvascular image-guided procedures + .REHLWHU &+8 +HQUL 0RQGRU 3DULV K K Pause cafĂŠ K K

Honorary Members - *RO]DULDQ HW + .REHLWHU

K K Sarcomaâ&#x20AC;&#x2122;s Imaging: what do, what to know ? 6 3DQ .8 /HXYHQ

AnnSymp2014.indd 1

K K Metastatic screening: from axial skeleton and bonesâ&#x20AC;Ś to whole body... and organe ) /HFRXYHW 8&/ %UX[HOOHV K K Walking Dinner K K The increasing role of economics in WKH PHGLFDO ÂżHOG / $QQHPDQV *HQW 8QLYHUVLW\ K K %HQHÂżW YHUVXV 5LVN LQ FDQFHU screening programs ' 7DFN &+ (SLFXUD %DXGRXU K K Training in Radiology in Europe - level 1,2,3 3URIHVVRU %LUJLW (UWO :DJQHU (65 (GXFDWLRQ &RPPLWWHH &KDLUPDQ

K K PICC and PAC: may I use them for CT/MRI exams and how to manage complications? 3 - %UX\qUH &+5 &LWDGHOOH /LqJH K K What is the role of Belmip and how important it is for radiologists? * 9LOOHLUV %65 3UHVLGHQW K K Closing remarks K K BSR General Assembly

)XOO QDPH $GGUHVV 7HOHSKRQH QXPEHU )D[ QXPEHU

Programme

REGISTRATION FORM (deadline October 31st, 2014) Annual Symposium Belgian Society of Radiology (BSR) Brussels, November 15th, 2014

OYR

RUG

ansb

eek

Military â&#x20AC;&#x201C;â&#x20AC;&#x201A; Hospital, Brussels 1â&#x20AC;&#x201A; â&#x20AC;&#x201C;â&#x20AC;&#x201A; 1120 Brussels Military Hospital, Brusselsâ&#x20AC;&#x201A; Rue Bruynstraat Rue Bruynstraat 1 E MME 1120 Registration information: denis.henroteaux@chrcitadelle.be ME AMME AMME AMME AMME AMME AMME MMERAMM MBrussels A A A R R R G GR ROGR ROGR ROGR ROGR ROGR G G G P P P P PRO PRO PRO P PRO PRO de R

Rue

e sb

Vilv o

ord

diologie PXVHXP adiologie

weg

teen

rasla

ses

de Ty

Militair Hospitaal Bruynstraat e Belge

Va nO

%XVLQHVV )DFXOW\

Beja r

eT yra

E19

E40

Av. de

Av .d

Koningslo Vilvoorde

nto

R0 E19

Av .A

Medialaan

iala

Med

21/08/14 08:26

JBR–BTR, 2014, 97: VI.

FORTHCOMING COURSES AND MEETINGS NATIONAL MEETINGS 25-26.09.14 Atelier de colonoscopie virtuelle Lecture en «Filet-View» Clinique Saint-Joseph, Liège Information: anne-marie.mandic@chc.be 02-04.10.14 HRCT of the Lung, Teaching course Leuven, Belgium Information: www.everyoneweb.com/chestradiology monika.philips@uzleuven.be 03.10.14 BSR – Cardiovascular and Interventional Radiology Mont-Godinne, UCL Information: Dr J.F. De Wispelaere jean-francois-dewispelaere@uclouvain.be

04.10.14 BSR – Neuroradiology Hotel Dolce La Hulpe, La Hulpe Information: neuroimagerie@chu.ulg.ac.be

13.11.14 BSR – Cardiac Imaging Woluwe, Sodehotel Information: nramollet@gmail.com

09.12.14 BSR – Pediatric Radiology Brussels, Iris Sud Information: mcassart@ulb.ac.be

12.12.14 BSR – Cardiovascular and Interventional Radiology Leuven, UZ Gasthuisberg Information: Dr G. Maleux geert.maleux@uzleuven.be 22-24.01.15 2ND LEUVEN COURSE ON HEAD AND NECK IMAGING: FROM SYMPTOM TO DIAGNOSIS Leuven, Belgium Information: www.headandneckimaging.be monika.philips@uzleuven.be 13.12.14 SEMINARS IN MSK RADIOLOGY Liège, University Hospital Information: www.chuliege.be/MSKradiology

BSR Annual Symposium 15.11.14

BSR – Neuroradiology 04.10.14

BSR – Cardiac Imaging 13.11.14

BSR – Pediatric Radiology 09.12.14

BSR-CVIR 03.10.14, 12.12.14

Miscellaneous 25-26.09.14, 02-04.10.14

Detailed and real time information is available on RBRS website at www.rbrs.org

InterNATIONAL MEETINGS

DIPLOME UNIVERSITAIRE D’IMAGERIE VASCULAIRE NON INVASIVE Année 2014-2015 Le Kremlin-Bicêtre, France Organisation: Pr M.F. Bellin, Pr P. Legmann Information : robin.menguy@bct.aphp.fr 13-17.09.14 CIRSE ANNUAL MEETING Glasgow, UK Information: www.cirse.org 17-19.09.14 EMRI – ABDOMINAL AND UROGENITAL Ankara, Turkey Information: walter.rijsselaere@uzbrussel.be 29.09-03.10.14 EMRI – MUSCULOSKELETAL Porto, Portugal Information: walter.rijsselaere@uzbrussel.be 02-04.10.14 Cardiac Imaging 2014 Paris, France Information: www.escr.org

25-Forthc. courses 97(4).indd 2

06-10.10.14 EMRI – CENTRAL NERVOUS SYSTEM II Riga, Latvia Information: walter.rijsselaere@uzbrussel.be 06-10.10.14 EMRI – BASIC MRI PHYSICS Madrid, Spain Information: walter.rijsselaere@uzbrussel.be 09-10.10.14 EMRI – CARDIOVASCULAR WITH CT CORRELATION Vienna, Austria Information: walter.rijsselaere@uzbrussel.be 16-20.10.14 JOURNEES FRANCAISES DE RADIOLOGIE Paris, France Information: www.sfrnet.org 07-08.11.14 ESSR Sports imaging Meeting Poland, Wroclaw Information: www.essr.org sudolszopinska@gmail.com maryam.shahabpour@uzbrussel.be

13-14.11.14 ESOI AUTUMN WORKSHOP 2014 Imaging cancer: staging and response to therapy Turin, Italy Information: www.esoi-society.org 13-25.11.14 7th EUROPEAN MULTIDISCIPLINARY COLORECTAL CANCER CONGRESS (EMCCC) Amsterdam, The Netherlands Information: www.dccg.nl/emccc2014 30.11-05.12.14 RSNA 2014 ANNUAL MEETING Chicago, USA Information: www.rsna.org 04-08.03.15 ECR 2015 Vienna, Austria Information: www.myesr.com

19/08/14 15:59

JBR–BTR, 2014, 97: 197-201.

PERCUTANEOUS CHOLECYSTOSTOMY: SINGLE CENTRE EXPERIENCE IN 111 PATIENTS WITH AN ACUTE CHOLECYSTITIS R. Peters1, S. Kolderman2, B. Peters3, M. Simoens4, S. Braak1 Purpose: To evaluate the safety and long-term outcome of percutaneous cholecystostomy (PC) under radiologic guidance for acute calculous cholecystitis (ACC) and acute acalculous cholecystitis (AAC) in all patients undergoing that procedure at our institution. Materials and methods: We performed a retrospective analysis of 111 patients who underwent PC from 2004 to 2012. Patients were divided into two groups: AAC and ACC. For all patients, comorbidity and American Society of Anesthesiologists (ASA) classification were determined. The indications, complications, recurrence rate and long-term outcome for both groups were analysed. The mean follow-up was 55 months. Results: Twenty-four patients with AAC and 87 patients with ACC underwent PC. The most common sonographic findings of ACC and AAC were gallbladder wall thickening (90,9%) and hydrops (72,9%). Twelve of 24 patients with AAC (50%) were hospitalized at the Intensive Care Unit (ICU). Overall, the procedure failed in 2 (1,8%) patients. There were 4 (3,6%) abscesses and 2 (1,8%) fistulas post PC. Drain dislodgment was found without sequelae in 8 (7,2%) patients. Elective cholecystectomy was performed in 35/111 (31,5%). Fifty-one of 87 (58,6%) patients with gallstones underwent cholecystectomy; 36/87 (41,3%) did not undergo surgery due to a too short follow-up or death of nonbiliary disease. In the AAC group, there was no recurrent cholecystitis in 17/24 (70,8%) patients; 3/24 (12,5%) underwent surgery and 4/24 (16,6%) patients died in the ICU. Conclusion: PC is a minimally invasive treatment with low complication rate for patients with acute cholecystitis whom considered being at high-risk for urgent cholecystectomy. Good selection (ASA III and IV) and indication is needed in patients with ACC before PC because the majority will be operated later on. AAC can be managed nonoperatively and further treatment might not be needed. Key-words: Gallbladder, interventional procedure – Cholecystitis.

Acute cholecystitis is a common cause of presentation at the emergency department. Acute inflammation of the gallbladder occurs mostly from persistent obstruction of the cystic duct or gallbladder neck by an impacted gallstone. In the minority of cases (5-10%), acute cholecystitis develops in a gallbladder without gallstones called AAC (1). AAC mainly occurs in seriously ill patients in the ICU or those who have recently undergone the stress of severe trauma or major surgery (2). The etiology is multifactorial and includes ischemia, gallbladder wall infection, chemical toxicity and cystic duct obstruction. Sonography is the preferred imaging modality for the initial evaluation of patients with suspected acute cholecystitis (1). False-positive results occur in the setting of gallbladder wall thickening in the absence of acute inflammation. Cirrhosis, congestive heart failure, ascites, hepatitis, gallbladder torsion or carcinoma can cause gallbladder wall thickening (3). Rarely, emphysematous cholecystitis is seen in patients with

diabetes in whom foci of gas are identified within the wall and / or lumen of the gallbladder (4). Surgery is the treatment of choice for acute cholecystitis and is typically performed at presentation if the duration of symptoms is less than 72 hours (5). There is a significant risk of increased operating time, higher conversion rate, more postoperative complications and mortality form urgent cholecystectomy in certain subgroups of patients such as the critically ill patient with comorbidity and in advanced stages of cholecystitis (15). Here, surgery can result in serious complications and even mortality. The mortality in lowrisk elderly patients is around 10%, but is as high as 46% in high-risk elderly patients (6, 7, 8). PC can be an alternative treatment in high risk patients. It allows immediate decompression of the acutely inflamed gallbladder (9, 10, 11). In our hospital, we regard PC as a useful treatment option for patients with high-risk or who present later than 72 hours and still have signs of hydropic, inflamed gallbladder on

From: 1. Department of Radiology, ZGT, Almelo, The Netherlands, 2. Department of Radiology, UMCG, Groningen, The Netherlands, 3. Department of Internal Medicine, AZ Monica, Antwerpen, Belgium, 4. Department of Surgery, ZGT Almelo, The Netherlands. Address for correspondence: Dr R. Peters, M.D., Department of Radiology, ZGT, Zilvermeeuw 1, 7609 PP Almelo, The Netherlands. E-mail: r_peters86@hotmail.com

peters-braak-.indd 197

ltrasound. The aim of this study u was to evaluate the safety success rate, ratio of delayed surgery, complications and clinical outcome of PC under radiologic guidance for ACC and AAC in all patients undergoing that procedure at our institution. Materials and methods We reviewed the medical records of 111 patients with acute cholecystitis who underwent PC in the Department of Radiology at Ziekenhuisgroep Twente (ZGT) Almelo/Hengelo in the Netherlands from January 2004 to January 2012. In all cases the diagnosis of acute cholecystitis was made based on clinical signs, laboratories and ultrasonography confirmation. Information entered included patient demographics, sonographic appearance, success rate of PC, drain dislodgement, complications and outcome. Patients were divided into two groups: AAC and ACC. All procedures were performed with ultrasound guidance by Seldinger technique. The gall bladder was visualized and local anesthetic infiltrated into the subcutaneous tissue. In all cases locking pigtail catheters (8.5-French Dawson-Mueller, Cook) were placed into the gallbladder using the free-hand trocar technique. A transhepatic approach, defined as the catheter tract traversing the liver parenchyma, was attempted when possible. Technical success was

5/08/14 13:15

198

efined as placement of a pigtail d catheter into the lumen of the gallbladder with aspiration of bile. The mean follow-up was 55 months. Patients A total of 111 patients were included (Table I). There were 93 patients with acute cholecystitis presenting at the emergency department and 18 hospitalized patients. Twelve of 18 inpatients were hospitalized at the ICU. There were 58 males and 53 females included with a median age of 72 years (range 25-93 years). Co-morbidity and American Society of Anesthesiologists (ASA) classification Most patients had significant ssociated conditions as listed in a Table II. Diabetes, cardiac or renal failure were the most frequent comorbidities in 88 of 111 patients with acute cholecystitis undergoing PC. Pulmonary comorbidities were mostly COPD related. Nine patients had a history of terminal malignancy and two polytrauma patients were hospitalized at the ICU. The American Society of Anaesthesiologists’ (ASA) classification was used as grading system for preoperative health of the patients. Patients of the ASA II group (9%) (primarily operable) had symptoms of cholecystitis occurring longer than 72 hours. Patients with severe systemic disease were 91% (ASA III and ASA IV) of the population (Table III). Sonographic appearance of acute cholecystitis Eighty-seven (78%) patients had calculous and 24 (22%) had acalculous cholecystitis (Table V). 87 patients presented with gallstones and in 24 cases there were no gallstones found. 81/111 (72,9%) patients with ACC presented at the Emergency Department. Twelve of 24 patients with AAC (50%) were hospitalized at the Intensive Care Unit. The most common sonographic finding of ACC and AAC was gallbladder wall thickening (90,9%) and hydrops (72,9%) (Table IV). Patients with AAC even presented with 91,6% and 95,6% respectively. The sonographic Murphy’s sign was positive in 50/111 (45,0%) patients (Fig. 1). Eight patients presented with perforation and abscess. Gas was present in 5 (4,5%) patients. A diagnostic CT was indicated for each complicated cholecystitis.

peters-braak-.indd 198

JBR–BTR, 2014, 97 (4)

Table I. — Age and gender distribution of patients with acute cholecystitis. Demography Age Mean Range Inpatients ED Total

Men

Women

Overall

72.6 35-93 12 42 58

71,5 25-92 6 51 53

72,1 25-93 18 93 111

Table II. — Comorbidities in 91 of 111 patients. Co-morbidities Cardiac failure Diabetes mellitus and renal failure Pulmonary CVA Malignancy Polytrauma

62 26 18 10 9 2

Table III. — ASA classifications of the patient population. ASA-classification ASA II ASA III ASA IV

Follow-up In 109/111 (98,2%) the placement of PC was successful. The cholecystostomy tube procedure failed in 2 (1,8%) patients because of pain and vasovagal reaction. The findings for complications were as follows: there were no procedure-related deaths, there were 4 (3,6%) abscesses and 2 (1,8%) fistulas post PC. Drain dislodgment was found without sequelae in 8 (7,2%) patients (Table VI). Twenty-four patients with AAC and 87 patients with ACC underwent PC. The mean follow-up was 55 months. In the AAC group, seventeen of 24 (70,8%) patients healed with PC and antibiotic treatment. None of these patients needed surgery in the follow-up period, so there was no recurrent cholecystitis. There were 2 patients of the AAC group who underwent emergency cholecystectomy and 1 elective surgery because the patient took the drain out. 4 patients died of other causes with their catheters in situ at the ICU. Fifty-one of 87 (58,6%) patients with gallstones underwent cholecystectomy at a later date; 34 (39%) patients recovered from the acute illness and underwent

9% 62% 29%

elective cholecystectomy. 17 (19,5%) patients needed an emergency cholecystectomy because of recurrent cholecystitis. 19/87 (21,8%) did not undergo surgery and there was no recurrent cholecystitis in the followup period. 17/87 (19,5%) died of nonbiliary cause mostly elderly patients with severe incurrent illnesses (Fig. 2). Overall, in 35/111 (31,5%) patients, PC was performed as a temporising measure, with final elective cholecystectomy at a later date. There were 36/111 (32,4%) patients presented with acute cholecystitis who had no recurrent cholecystitis. Nineteen (19/111) patients needed emergency cholecystectomy (Table VI). Discussion Sonography is the imaging modality of choice for the initial evaluation of patients with suspected acute cholecystitis. Most sonographic findings are non-specific but are suggestive for acute cholecystitis (1, 3). It is important to demonstrate gallstones in the gallbladder. Ultrasound can detect stones and has a sensitivity of approximately 95% and a specificity

5/08/14 13:15

PERCUTANEOUS CHOLECYSTOSTOMY — PETERS et al

199

Table IV. — Sonographic findings.

CALCULOUS (n = 87)

ACALCULOUS (n = 24)

TOTAAL (n = 111)

79 (90,8%) 58 (66,6%) 35 (40,2%) 35 (40,2%) 21 (24,1%) 7 (8,0%) 7 (8,0%) 3 (3,4%)

22 (91,6%) 23 (95,6%) 15 (62,5%) 12 (50%) 9 (37,5%) 1 (4,1%) 1 (4,1%) 2 (8,3%)

101 (90,9%) 81 (72,9%) 50 (45,0 %) 47 (42,3%) 30 (27%) 8 (7,2%) 8 (7,2%) 5 (4,5%)

Gallbladder wall thickening Hydrops Murphy’s sign Sludge Pericholecystic fluid Perforation Abscess Gas

Fig. 1. — Sonographic findings

Table V. — Acute calculous and acalculous cholecystitis. Inpatients ED IC Totaal

Calculous

Acalculous

Overall

3 81 3 87

3 12 9 24

6 93 12 111

of appr. 97% (12). In our study, gallbladder wall thickening and hydrops are the most common sonographic appearance for ACC and AAC. Murphy sign was in 45% positive but we believe our patient group – mostly elders and IC patients – were less reliable for pain stimulus. ACC is more common in patients with acute cholecystitis diagnosed at the ED, whereas AAC is mainly found in the

peters-braak-.indd 199

critically ill, already hospitalised patient. Ultrasonography remains operator dependent. In this study PC was performed for critically ill patients who are not acceptable candidates for emergency surgery because of severe comorbidities and in advanced stages of cholecystitis. The general medical condition of our PC patients is a reflection of the older age and a high

prevalence of comorbidities, so these patients are categorised with a high ASA classification. In the present series, the minority group were ASA II patients (9%) who have been symptomatic for more than 72 hours and the surgery decided not to operate. 91% of our 111 patients were classified as ASA III and IV. ASA III and IV patients have a too high surgical or anaesthetic risk for urgent cholecystectomy. Tube cholecystostomy is a procedure to decompress the acutely inflamed gallbladder. Successful drainage by PC was achieved in 98,2% of the patients. The procedure failed in 2 patients because of pain and vasovagal reaction. This result is consistent with the literature (13). So, the technically success rate is very high in experienced hands. The access route can be either transperitoneal (TP) or transhepatic (TH). In all patients, PC was performed under local anaesthesia with ultrasound guidance. Unfortunately, in our data there was a lack of description of the access route and comparison of complications or drain dislodgements for TH or TP could not be made. Potential disadvantages of using the TP route are an increased risk of bile peritonitis and colon perforation. The gallbladder could also be more difficult to puncture due to gallbladder mobility. The TP has a decreased risk of bleeding and secondary liver contamination by infected bile. The TH approach decreases the risk of bile leak, portal vessel injury and colon perforation. However, it carries a risk of pneumothorax and bleeding from the liver parenchyma (11, 15). In the current literature there is still debate as to whether which route is preferable. Generally, the radiologists choose the shortest path but the procedure remains interventional radiologist dependent. Technical problems with cholecystostomies are few. There were 8 drain dislodgements and these patients were managed conservative

5/08/14 13:15

200

JBR–BTR, 2014, 97 (4)

Table VI. — Follow-up, complication and technical success rate. No recurrence Elective lap. Chol. Emergency Chol. Dead in 6 month Complication Drain dislodgement Procedure fail

Calculous

Acalculous

Total

19/87 (21,8%) 34/87 (39,0%) 17/87 (19,5%) 17/87 (19,5%) 4/87 (4,5%) 5/87 (5,7%) 2/87 (2,2%)

17/24 (70,8%) 1/24 (4,1%) 2/24 (8,3%) 4/24 (16,6%) 2/24 (8,3%) 3/24 (12,5%) 0/24 (0%)

36/111 (32,4%) 35/111 (31,5%) 19/111 (17,1%) 21/111 (18,9%) 6/111 (5,4%) 8/111 (7,2%) 2/111 (1,8%)

Conclusion In our experience, PC is a safe intervention technique with a high success rate. The complication rate for PC is very low and our results correlated with previous studies. PC is a valuable technique for the management of patients with acute calculous or acalculous cholecystitis. Once the acute symptoms resolve elective cholecystectomy should be followed for patients with gallstones. References

Fig. 2. — Outcome

without complications. The risk of tube dislodgement can be minimized by secure fixation to the skin and use of self-retaining loop catheters. In our study there were no procedure-related deaths. There were 4 abscesses and 2 fistulas post PC. The overall frequency was very low (5,4%). The patients had non-fatal complications and were treated conservatively. During the follow-up period, 70% of the patients with AAC were treated with PC and had no recurrence and needed no surgery later on. Only 3 interval cholecystectomies have been performed whom 2 for recurrence cholecystitis during long-term follow-up. Therefore, PC may provide definitive treatment in the management of AAC and the majority of patients do not require subsequent cholecystectomy. 58,6% of patients with ACC were operated later on. Thirty-four patients recov-

peters-braak-.indd 200

ered from the acute illness and underwent elective cholecystectomy. Seventeen patients needed an emergency cholecystectomy because of recurrent cholecystitis. PC is an effective treatment in ACC to decompress the inflamed gallbladder but a definitive surgical treatment has to be well-timed planned. Our data are comparable to results reported by the systematic review conducted by Winbladh et al. (13). Many authors confirm the safety and effectiveness of cholecystostomy for the initial treatment (7, 8, 9). Proper patient selection is important in the ACC group to reduce the risk of recurrence and to avoid urgent cholecystectomy (11, 14, 16). There are many controversies in the current literature. Guidelines with regard to the optimal timing of surgical intervention and management have to be made in treatment of ACC.

1. Hanbidge A., Buckler P., et al.: Imaging evaluation for acute pain in the right upper quadrant. RadioGraphics, 2004, 24: 1117-1135. 2. Shapiro M.J., Luchtefeld W.B., Kurzweil S., et al.: Acute acalculous cholecystitis in the critically ill. Am Surg, 1994, 60: 335. 3. Gore R.M., Yaghmai V., Newmark G.M., Berlin J.W., Miller F.H.: Imaging of benign and malignant disease of the gallbladder. Radiol Clin N Am, 2002, 40: 1307-1323. 4. Garcia-Sancho T., Fernandez de Lis J.A., et al.: Acute emphysematous cholecystitis. Report of twenty cases. Hepatogastroenterology, 1999, 46: 2144. 5. Morse B.C., Smith J.B., Lawdahi R.B., Roettger R.H.: Management of acute cholecystitis in critically ill patients: contemporary role for cholecystostomy and subsequent cholecystectomy. Am Surg, 2010, 76: 708. 6. Pessaux P., Regenet N., Tuech J., et al.: Laparoscopic versus open cholecystectomy: a prospective comparative study in elderly with acute cholecystitis. Surg Laparosc Endosc Percutan Tech, 2001, 11: 252-255. 7. Avrahami R., Badani E., W atemberg S., et al.: The role of percutaneous transhepatic cholecystostomy in the management of acute cholecystitis in high-risk patients. Int Surg, 1995, 80: 111-114. 8. Byrne F., Suhocki P., Mitchell R.: Percutaneous cholecystostomy in patients with acute cholecystitis: Experience of 45 patients at a US referral center. J Am Coll Surg, 2003, 197: 206-211.

5/08/14 13:15

PERCUTANEOUS CHOLECYSTOSTOMY — PETERS et al

9. Joseph T., Unver K., et al.: Percutaneous Cholecystostomy for Acute Cholecystitis: Ten-Year Experience. JVIR, 2001, 23: 83-88.e1. 10. Ito K., Fujita N., Noda Y.: Percutaneous cholecystostomy versus gallbladder aspiration for acute cholecystitis: a prospective randomized controlled trial. AJR Am J Roentgenol, 2004, 183: 193. 11. Basaran O., Yavuzer N., et al.: Ultrasound-guided percutaneous cholecystotomy for acute cholecystitis in critically ill patients: Once center’s

experience. Turk J Gastroenterol, 2005, 16: 134-137. 12. Shea J., Berlin A., Escarce J.: Revised estimates of diagnostic test sensitivity and specificity in suspected biliary tract disease. Arch Intern Med, 1994, 154:2573-2581. 13. Winbladh A., Gullstrand P., Svanvik J., Sandstrom P.: Systematic review of cholecystostomy as a treatment option in acute cholecystitis. HPB (Oxford) 2009, 11: 183-193. 14. Patel M., Miedema B.W., James M.A., et al.: Percutaneous cholecystostomy

201

is an effective treatment for high-risk patients with acute cholecystitis. Am Surg, 2000, 66: 33-37. 15. Kaufman J.A., Lee M.J.: Gallbladder intervention. In: Kaufman JA, Lee MJ Vascular & interventional radiology: The Requisites. Philadelphia, 2004, pp 588-601. 16. McGillicuddy E.A., Schuster K.M., Barre K., et al.: Non-operative management of acute cholecystitis in the elderly. Br J Surg, 2012, 99: 12541261.

ANNOUNCEMENT

Engage:

Chef de Service – Radiologie (H/F) Site Godinne Recrutement interne – externe

Le CHU Dinant Godinne compte 640 lits justifiés et représente, grâce à ses 2300 travailleurs et ses 350 médecins, le premier employeur privé de la Province de Namur & du Luxembourg. La mission de notre Institution est de dispenser des soins d’excellente qualité pour tous, tant sur le plan humain que médical et développer la recherche, l’enseignement et les services qui les optimisent. De plus, il compte également deux crèches, des lits MR-MRS et est impliqué dans plusieurs ASBL périphériques. Plus précisément, ce service comprend:

2 CT de dernière génération (Reconstruction Itérative de Modèle), 2 IRM (1.5 et 3 T), Pet-CT, Spect-CT (collaborations avec la médecine nucléaire), Angiographie et radiologie interventionnelle, Différents pôles: sénologie, thoraco-abdominal, neuroradiologie, ostéo-articulaire et radiologie conven tionnelle, ➢ 12 médecins et 6 assistants.

➢ ➢ ➢ ➢ ➢

Conditions du poste:

• Statut Salarié • Temps plein Activités principales:

• • • •

Activités cliniques de radiologie Missions universitaires Assurer la gestion et la dynamique d’une équipe Participer à des projets institutionnels

Profil recherché:

• Médecin spécialiste en Radiologie • Adhésion aux valeurs institutionnelles Renseignements: 081/42.30.49 • Professeur Yves BOUTSEN, Directeur Médical Adjoint Cellule Recrutement & Sélection: 081/42.28.04 • Modalités d’introduction des candidatures: Cv & lettre de motivation à envoyer avant le 15 Septembre 2014 à: recrutement-chu@uclouvain.be

peters-braak-.indd 201

14/08/14 12:58

JBR–BTR, 2014, 97: 202-205.

How sensitive and specific are MRI features of sacroiliitis for diagnosis of spondyloarthritis in patients with inflammatory back pain? L. Jans1, L. Coeman1, L. Van Praet2, P. Carron2, D. Elewaut2, F. Van den Bosch2, J.L. Jaremko3, W. Huysse1, K.L. Verstraete1 Objective: To determine the sensitivity and specificity of MRI features of sacroiliitis in spondyloarthritis (SpA). Materials and methods: A retrospective study reviewed MRI of the sacroiliac (SI) joints in 517 patients with inflammatory back pain. Sensitivity, specificity, positive and negative likelihood ratios of active and structural lesions of sacroiliitis with final clinical diagnosis as golden standard was calculated. Results: MRI showed active inflammation in 42% of patients (bone marrow oedema (BMO) (41.5%), capsulitis (3.3%), enthesitis (2.5%)) and structural changes in 48.8% of patients (erosion (25%), fat infiltration (31.6%), sclerosis (32%) and ankylosis (7.6%)). BMO was the MRI feature with the highest sensitivity (65.1%) for diagnosis of SpA. Capsulitis (99%), enthesitis (98.4%), ankylosis (97.4%) and erosion (94.8%) had a high specificity for diagnosis of SpA, whereas BMO (74.3%), sclerosis (75.8%) and fat infiltration (84.0%) were less specific. BMO concomitant with enthesitis, capsulitis or erosions increased the specificity. Concomitant presence of BMO and sclerosis or fat infiltration decreased the specificity. Conclusion: BMO is moderately sensitive and specific for diagnosis of SpA in patients with inflammatory back pain. BMO concomitant with enthesitis, capsulitis, ankylosis or erosion increases the specificity. Concomitant fat infiltration or sclerosis decreases the specificity for diagnosis of SpA. Of all lesions, erosion had by far the highest positive likelihood ratio for diagnosis of SpA. Keywords: Spine, MR – Arthritis.

Spondyloarthritis (SpA) is a group of inflammatory joint conditions sharing common clinical, radiological, genetic and even therapeutic characteristics and are often associated with the presence of human leukocyte antigen (HLA)-B27 (1-5). Early diagnosis of SpA has gained significance for rheumatologists as new medical treatment options have become available (6). MRI of the SI joints shows active inflammatory and structural lesions of sacroiliitis long before radiographic changes become evident (7, 8). Active sacroiliitis on MRI is an important classification criterion. MRI is regarded ‘positive’ for sacroiliitis if bone marrow oedema (BMO) is clearly present (8). Other MRI features representing active inflammation of the SI joint such as enthesitis or capsulitis alone are not sufficient for a ‘positive’ MRI for sacroiliitis. Structural lesions in sacroiliitis are sclerosis, fat infiltration, erosion and finally ankylosis (8). These active and structural changes of the SI joints, in particular the presence of BMO, may also be presentin patients presenting with non-rheumatological entities that clinically mimic sacroiliitis such as degenerative disease, lumbosacral

transitional anomaly, spondylolysis, fracture, infection and tumor (9, 10). The aim of this study is to determine the sensitivity and specificity of MRI features of sacroiliitis in SpA. Also, we sought to find if BMO concomitant with other MRI features of sacroiliitis may increase the sensitivity and specificity for diagnosis of SpA. Materials and methods The retrospective study was approved by the institutional ethics committee. Study group All participants, aged 16-45 years old, were recruited from the hospital rheumatology outpatient clinics in a tertiary care center and were referred for MRI of the SI joints with clinical suspicion of sacroiliitis. Clinical criteria for ‘inflammatory type’ back pain included a. age at onset < 40 years, b. insidious onset, c. improvement with exercise, d. no improvement with rest, e. pain at night (8). Patients who underwent back surgery were excluded from the study. From March 2005 to February 2013, 517 patients were included in

From: 1. Department of 1. Radiology and Medical Imaging, 2. Rheumatology, Ghent University Hospital, Ghent, Belgium, 3. Department of Radiology, University of Alberta Hospital, Edmonton, Alberta, Canada. Address for correspondence: Dr L. Jans, M.D., Ph.D., Dpt of Radiology, Ghent University Hospital, De Pintelaan 185, 9000 Gent, Belgium. E-mail: lennartjans@hotmail.com

Jans et al.indd 202

the study (185 (35.8%) men, 332 (64.2%) women), with a median age of 33.3 years (range 16.1-44.9). We recorded from the clinical files if patients fulfilled the ASAS (Assessment of SpondyloArthritis international Society) classification criteria for axial or peripheral SpA (8). 210 patients (114 women, 96 men), with a median age of 30.8 years (range 16.1-44.9) were classified to have SpA (89.5% axial SpA, 10.5% peripheral SpA). The ASAS classification of these patients was considered the gold standard (8). 307 patients were not diagnosed with SpA (89 (29%) men, 218 (71%) women), with a median age of 34.7 years (range 16.2-44.9). In this group, MRI findings were normal in 211 patients. In 96 patients non rheumatologic findings were present: L5-S1 degenerative changes in 57 patients (60.6%), lumbosacral transitional anom aly in 25 patients (4.8%), hip joint disease in 10 patients (1.9%), osteitis condensans ilii in 12 patients (2.3%), DISH in 3 patients (0.6%), tumor in 5 patients (1%), fracture in 4 patients (0.8%) and infection in 1 patient (0.2%). Magnetic resonance imaging MRI was performed on a 1.5 T MRI unit (Avanto/Symphony, Siemens Medical, Erlangen, Germany). The SI joints were imaged in a body flexed array coil (Siemens Medical, Erlangen, Germany). Sequence protocol included: semicoronal (along long

5/08/14 13:18

MRI FEATURES OF SACROILIITIS FOR DIAGNOSIS OF SPONDYLOARTHRITIS — JANS et al

axis of the sacral bone perpendicular to the S2 vertebral body) T1-weighted turbo spin echo (TSE) (slice thickness (ST): 3 mm; repetition time/ echo time (TR/TE): 595/20 ms); semicoronal STIR (ST: 3 mm; TR/TE/TI: 5030/67/150 ms); axial fat-saturated (FS) T2-weighted TSE (ST: 4 mm; TR/TE: 4000/77 ms); axial STIR (ST: 5 mm; TR/TE/TI: 7540/67/150 ms). As per ASAS guidelines, no contrastenhanced pulse sequences were obtained (8). Image review The MR images were reviewed for the presence of active or structural lesions of sacroiliitis by 2 musculoskeletal radiologists with 10 and 14 years of experience (LJ,WH), who were blinded to clinical and other imaging findings. – Active lesions of sacroiliitis included BMO, enthesitis and capsulitis (8). BMO was defined as ‘positive’ for sacroiliitis if high T2 FS/STIR signal of the ilium or sacrum typically located periarticularly was present. If there is one signal (lesion) only, this should be present on at least two slices. If there is more than one signal on a single slice, one slice was considered to be enough (8). Enthesitis was defined high T2 FS/STIR signal of an enthesis representing BMO, soft tissue inflammation or joint/bursal fluid. Capsulitis is seen as high signal on STIR images involving the anterior or posterior capsule of the SI joint (7, 8). – Structural lesions of sacroiliitis consisted of sclerosis, fat infiltration, erosion and ankylosis. Sclerosis is depicted as low signal subchondral bands extending at least 5 mm by all sequences. Erosionsare low T1 signal bone defects at the joint margin that may occur throughout the cartilaginous joint compartment. Fat deposition depicts as periarticular high T1 signal in the bone. Ankylosis of the SI joint appears as low signal by all sequences bridging the SI joint and may show high T1 signal if bone marrow is present (8). Representative images are presented in Figs. 1-6. Statistical analysis Statistical analysis was performed using software package SPSS 20.0 for Windows (SPSS, Chicago, IL, USA). Basic descriptive statistics were performed where appropriate.

Jans et al.indd 203

203

A Fig. 1. — BMO and capsulitis of the SI joint in a 22-year-old male with spondyloarthritis. Semicoronal STIR image shows BMO (arrows) of the left SI joint with concomitant posterior capsulitis (short arrows).

B Fig. 2. — BMO and enthesitis of the SI joint in a 24-year-old female with arthritis. (A-B) Semicoronal spondylo STIR images show BMO (arrows) of the right SI joint with concomitant enthesitis of the left retroarticular ligaments (long arrow).

Diagnostic utility of active and structural lesions of sacroiliitis for diagnosis of SpA were determined by calculating sensitivity, specificity, positive and negative likelihood ratio for consensus reader data with final clinical diagnosis as golden standard.

Fig. 3. — BMO and concomitant erosion of the SI joint in a 26-year-old female with spondyloarthritis. A. Semicoronal T1-weighted MR image shows extensive erosions of the SI joints (arrows). B. Semicoronal STIR image shows BMO of both SI joints (arrows).

Active MRI lesions in sacroiliitis

Fig. 4. — BMO and ankylosis of the SI joint in a 33-year-old male with spondyloarthritis. Semicoronal (A) T1-weighted and (B) STIR MR images show ankylosis of the left SI joint (short arrows) with concomitant BMO of the right SI joint (long arrows).

MRI showed active lesions in 42% of patients (bone marrow oedema (BMO) (41.5%), capsulitis (3.3%), enthesitis (2.5%). BMO was the MRI feature with the highest sensitivity (65.2%) for diagnosis of SpA, with a

specificity of 74.6%. Presence of enthesitis and capsulitis had a low sensitivity, but a very high specificity for diagnosis of SpA.

Results

5/08/14 13:18

204

JBR–BTR, 2014, 97 (4)

Structural MRI lesions in sacroiliitis MRI showed structural lesions in 48.8%. Erosion had a moderate sensitivity but high specificity. Ankylosis had a low sensitivity but very high specificity for diagnosis of SpA. Sclerosis and fat infiltration had moderate sensitivity but lower specificity compared to ankylosis and erosion. Of all lesions, the presence of erosion had the highest LR+ (10.4) for diagnosis of SpA (Table I).

Diagnostic utility of lesions in sacroiliitis

Fig. 5. — BMO and fat infiltration of the SI joint in a 44-year-old female with degenerative joint changes.Semicoronal (A) T1-weighted and (B) STIR MR images show fat infiltration of the SI joints (short arrows) with concomitant BMO of the left SI joint (long arrow).

Fig. 6. — BMO and sclerosis of the SI joint in a 44-year-old female with degenerative joint changes. Semicoronal (A) T1-weighted and (B) STIR MR images show sclerosis of the right SI joint (short arrows) with concomitant BMO of both SI joints (long arrows).

Table I. — The sensitivity, specificity, positive and negative likelihood ratios of MRI features of sacroiliitis for the diagnosis of SpA. N ACTIVE BMO Enthesitis Capsulitis STRUCTURAL Sclerosis Fat infiltration Erosion Ankylosis

Sensitivity % Specificity %

LR+

LR-

215 13 17

65.2 3.8 6.7

74.6 98.4 99.0

2.56 2.38 6.7

0.47 0.98 0.94

167 164 130 39

43.3 54.8 54.3 14.8

75.6 84.0 94.8 97.4

1.77 3.42 10.4 5.7

0.75 0.54 0.48 0.87

(N = number of patients; LR+ = positive likelihood ratio; LR- = negative likelihood ratio; BMO = bone marrow oedema).

Table II. — The sensitivity, specificity, positive and negative likelihood ratios of BMO concomitant with other MRI features of sacroiliitis for the diagnosis of SpA. BMO + Enthesitis + Capsulitis + Sclerosis + Fat infiltration + Erosion + Ankylosis

N 215 10 17 109 112 120 26

Sensitivity % Specificity % 5.1 10.2 57.0 67.9 76.6 16.8

96.1 96.1 60.3 75.6 80.8 96.1

LR+

LR-

1.31 2.67 1.43 2.78 3.99 4.31

0.99 0.93 0.71 0.42 0.29 0.87

(N = number of patients; BMO = bone marrow oedema; LR+ = positive likelihood ratio; LR- = negative likelihood ratio).

Jans et al.indd 204

Capsulitis (99%), enthesitis (98.4%), ankylosis (97.4%) and erosion (94.8%) had a high specificity for diagnosis of SpA. Fat infiltration (84.0%), sclerosis (75.6%) and BMO (74.6%) were less specific. BMO concomitant with enthesitis, capsulitis or erosions increased the specificity. Concomitant presence of sclerosis or fat infiltration decreased the specificity. BMO concomitant with ankylosis, erosion and fat infiltration had a moderate higher positive likelihood ratio (LR+) for diagnosis of SpA, compared to the LR+ of concomitant presence of enthesitis and sclerosis (Table II). Discussion Early assessment of inflammation in the course of SpA gains importance in the light of new therapeutic strategies. The value of MRI of the SI joints is well established and resulted in a definition of a ‘positive MRI’ for sacroiliitis (8, 11, 12). In the current ASAS criteria of axial SpA MRI plays an important role: a ‘positive MRI’ is a key criterion for disease classification (8, 13, 14). However, in daily radiology practice it remains challenging to decide if demonstrated BMO is sufficient to really represent active sacroiliitis as seen in SpA. In our study we found a moderate sensitivity (65%) and -in this context more importantly- only a moderate specificity (74%) of BMO of the SI joints for diagnosis of SpA. These figures are similar to the figures published in the paper by Weber et al., who also reported on the limitations of using BMO as a single criterion in the current ASAS definition of a ‘positive’ MRI (16). We confirmedtheir findings, and also showed that concomitant fat infil tration and sclerosis decrease the diagnosticvalue of MRI. This is not surprising, since these features are commonly seen in degeneration,

5/08/14 13:18

MRI FEATURES OF SACROILIITIS FOR DIAGNOSIS OF SPONDYLOARTHRITIS — JANS et al

with mechanical back pain mimicking inflammation in these patients (15). Our study showed that the concomitant presence of other features of active sacroiliitis such as enthesitis or capsulitis, indicating an ongoing true inflammatory process, increased the specificity. The presence of structural lesions also contributes substantially to the diagnostic utility of MRI for diagnosis of SpA. The concomitant presence of erosions and ankylosis – both hallmarks of the disease – also increased the specificity (8). On the other hand, the concomitant presence of fat infiltration and sclerosis decreased the specificity for diagnosis of SpA, which may be explained by the presence of the same MRI features in degenerative processes (15). Weber et al stressed the importance of erosions as an MRI feature of SpA (16-17). In our study we found that of all lesions of the SI joints, the presence of erosion had the highest LR+ (10.4) for diagnosis of SpA. This finding confirms that erosion could play a role as a new or additional criterionin future classification systems, especially as it improves the specificity of MRI of the SI joints for diagnosis of SpA. In our study we also found a very high specificity of enthesitis (98%) and capsulitis (99%) for diagnosis of SpA. However, as these lesion were not commonly seen (in 4% and 6% respectively), they might not be as useful as classification criteria. Still, the presence of enthesitis or capsulitis may be particularly helpful in equivocal cases or may indicate that active inflammation is present at a certain stage in the disease process. There are some limitations to our study. Firstly, MRI was the only imaging technique, without correlation with radiography or CT. Secondly, the patient population represented referrals from a single tertiary center; referral patterns for sacroiliitis

Jans et al.indd 205

may vary elsewhere. This will particularly affect the reported likelihood ratios. Thirdly, as MRI is included as a criterion in the current ASAS classification, there is a risk of circular reasoning. Finally, we only studied patients with inflammatory back pain clinically suspected of SpA, no control group of age and sex-matched individuals was studied for comparison. In conclusion, we found that BMO is an important MRI feature of sacroiliitis with a moderate sensitivity and specificity for diagnosis of SpA in patients with inflammatory back pain. The specificity increases if concomitant enthesitis, capsulitis, erosions or ankylosis is present, but decreases if concomitant fat infiltration and sclerosis is demonstrated. Of all lesions, erosion has by far the highest positive likelihood ratio for diagnosis of SpA. References 1. Braun J., Sieper J.: Ankylosing spondylitis. Lancet, 2007, 369: 1379-1390. 2. Lacout A., Rousselin B., Pelage J.: CT and MRI of spine and sacroiliac involvement in spondylarthropathy. Am J Roentgenol, 2008, 191: 10161023. 3. Dougados M., Van der Linden S., Juhlin R., et al.: The European Spondylarthropathy Study Group preliminary criteria for the classification of spondylarthropathy. Arthritis Rheum 1991, 34:1218-1227. 4. Dougados M., Baeten D.: Spondyloarthritis. Lancet, 2011, 377: 2127-2137. 5. Mager A.K., Althoff C.E., Sieper J.: Role of whole-body magnetic resonance imaging in diagnosing early spondyloarthritis. Eur J Radiol, 2009 71: 182-188. 6. Lambert R.G., Salonen D., Rahman P., et al.: Adalimumab significantly reduces both spinal and sacroiliac joint inflammation in patients with ankylosing spondylitis: a multicenter, randomized, double-blind, placebo-controlled study. Arthritis Rheum, 2007, 56: 4005-4014.

205

7. Eshed I., Bollow M., McGonagle D.G., et al.: MRI of enthesitis of the appendicular skeleton in spondyloarthritis. Ann Rheum Dis, 2007, 66: 1553-1559. 8. Sieper J., Rudwaleit M., Baraliakos X.: The Assessment of SpondyloArthritis international Society (ASAS) handbook: a guide to assess spondyloarthritis. Ann Rheum Dis, 2009, 68:ii1ii44. 9. Bereau M., Prati C., Wendling D.: Sacroiliac edema by MRI does not always indicate spondylarthritis. Joint Bone Spine, 2011, 78: 646. 10. Schueller-Wiedekamm C., Schueller G.: Sacroiliitis oder pseudosacroiliitis? Radiologe, 2012, 52: 132-140. 11. Weber U., Østergaard M., Lambert R.G.: The impact of MRI on the clinical management of inflammatory arthritides. Skeletal Radiol, 2011, 40: 11531173. 12. Aydin S.Z., Maksymowych W.P., Bennett A.N., et al.: Validation of the ASAS criteria and definition of a positive MRI of the sacroiliac joint in an inception cohort of axial spondyloarthritis followed up for 8 years. Ann Rheum Dis, 2011, 71: 56-60. 13. Rudwaleit M., van der Heijde D., Landawé R., et al.: The Assessment of SpondyloArthritis international Societyclassification criteria for peripheralspondyloarthritis and for spondyloarthritis in general. Ann Rheum Dis, 2011, 70: 25-31. 14. Rudwaleit M., Jurik A.G., Hermann K.G., et al.: Defining active sacroiliitis on magnetic resonance imaging (MRI) for classification of axial spondyloarthritis: a consensual approach by the ASAS/OMERACT MRI group. Ann Rheum Dis, 2009, 68: 1520-1527. 15. Maus T.: Imaging the back pain patient. Phys Med Rehabil Clin N Am, 2012, 21: 725-66. 16. Weber U., Maksymowych W.P.: Sensitivity and specificity of magnetic resonance imaging for axial spondyloarthritis. Am J Med Sci, 2011, 341: 272277. 17. Weber U., Lambert R.G., Østergaard M., Hodler J., Pedersen S.J., Maksymowych W.P.: The diagnostic utility of magnetic resonance imaging in spondylarthritis: an international multicenter evaluation of one hundred eighty-seven subjects. Arthritis Rheum, 2011, 62: 3048-3058.

5/08/14 13:18

JBR–BTR, 2014, 97: 206-210.

REAL TIME ULTRASOUND GUIDED PEDIATRIC PERCUTANEOUS RENAL BIOPSY: THE TRADITIONAL METHOD VERSUS ANGLED TANGENTIAL APPROACH K.C. Caliskan¹, G. Ozcelik², E. Cakmakci¹, S. M. Ulusay¹, A.S. Celebi¹, S. Turk³, A. Ozagari4, Z. Karpat¹ Aim: The aim of the present study was to compare the success and complication rates of pediatric renal biopsy procedures between the angled tangential approach and the traditional approach. Methods: From 2004 to 2009 we prospectively enrolled pediatric patients who had undergone real time ultrasound guided renal biopsy with angled tangential approach. For comparison, we retrospectively reviewed pediatric patients who had undergone traditional renal biopsy between 2002 and 2004. Adequacy of renal tissue histopathological samples and the complication rates were compared between groups. Results: One hundred twenty-eight patients underwent traditional renal biopsy (Group A) while 166 patients underwent biopsy performed with angled tangential approach (Group B). The rate of inadequate material was higher in Group A compared to Group B (6.3% vs. 0.6%, p = 0.006). In four cases (three in Group A and one in Group B) renal biopsies revealed normal renal tissue. While a major complication (hemoperitoneum requiring transfusion) occurred in one case in Group A, no major complications were seen in Group B. Conclusion: Compared with the traditional technique, the angled tangential approach resulted in a higher adequate material rate and lower complication rate.These findings indicate that angled tangential approach could be considered for pediatric percutaneous renal biopsies.

JA KLEUR

Key-words: Kidney, biopsy – Ultrasound (US, guidance).

Percutaneous renal biopsy (PRB) is a routine method for diagnosing parenchymal diseases since it was first carried out in 1951 in parallel to the development of imaging tools (14). Renal biopsies, which performed under the guidance of direct urinary system and intravenous pyelography graphies previously were replaced by real time biopsies under the guidance of ultrasonography (US). Indications for PRB in children include persistent proteinuria and/or hematuria of unknown origin, differentiation of nephrotic syndrome and/or nephritic syndrome, rapidly progressive glomerulonephritis, and acute or chronic renal failure when the cause and extent of the disease are unknown. In patients with systemic disorders such as systemic lupus erythematosus or Henoch-Schönlein purpura, examination of renal tissue may be necessary to document the histological type and magnitude of the renal injury which is necessary in planning management of the disease (3, 4). PRB is a safe and practical procedure that provides important information to the pediatric nephrologist in establishing the diagnosis, evaluating the acuteness and severity of the disease, monitoring disease

Fig. 1. — Corticomedullar position of biopsy needle for obtaining material to angled tangential approach on VRT (Volume Rendering Technique) image.

progression, and assessing the response to therapy (1-4). Its efficiency increased with routine practice and with the experience obtained. Although the rate of complications decreased considerably, it still has major and minor complications that have to be taken into consideration, being an invasive procedure. In this study, we aimed to compare the success and complication

From: 1. Department of Radiology, Sisli Etfal Training and Research Hospital, Istanbul, Turkey, 2. Department of Pediatrics, Division of Pediatric Nephrology, Sisli Etfal Training and Research Hospital, Istanbul, Turkey, 3. Department of Anesthesia and Reanimation, Sisli Etfal Training and Research Hospital, Istanbul, Turkey, 4. Department of Pathology, Sisli Etfal Training and Research Hospital, Istanbul, Turkey. Address for correspondence: Dr Emin Cakmakci, M.D., Department of Radiology, Sisli Etfal Training and Research Hospital, Istanbul, Turkey. E-mail: em_sel74@hotmail.com

Caliskan et al.indd 206

rates of pediatric renal biopsy procedures between the angled tangential approach and the traditional approach. Material and methods Study design We retrospectively reviewed pediatric patients who had undergone traditional renal biopsy between March 2002 and May 2004 (Group A). For comparison, we prospectively enrolled consecutive pediatric patients who had undergone real time ultrasound guided renal biopsy with angled tangential approach from June 2004 to May 2009 (Group B). Ethical Committee approval was obtained.

5/08/14 13:33

REAL TIME ULTRASOUND GUIDED PEDIATRIC PERCUTANEOUS RENAL BIOPSY — CALISKAN et al

207

Fig. 2. — Biopsy needle route with angle between 45-60 degrees with reference to the renal capsule or base line of the convex probe.

The study protocol conformed to the Helsinki declaration; all patients (parents) were fully informed and signed consent forms. After obtaining informed consent, subjects were screened with systemic arterial blood pressure, complete blood count, coagulation parameters, and urine analysis. Cases with hypertension underwent the procedure after being rendered normotensive. Cases with impaired coagulation parameters underwent the procedure after parameters were corrected. In addition, cases with active urinary system infection underwent biopsy after their infection was controlled. Nephrotic syndrome refractory to steroids, nephritic syndrome, hematurea and proteinurea, acute or chronic renal failure whose cause is unknown and were considered to be

Caliskan et al.indd 207

indicated for biopsy were included in our study. Biopsy indication was determined by pediatric nephrology clinic in all cases. Percutaneous renal biopsy procedure All procedures were performed by two experienced interventional radiologists who had performed at least 400 renal biopsies prior to this study. The biopsies were performed under sedation with midazolam (0.03 mg/ kg, IV), ketamine (1 mg/kg, IV) and locally administrated prilocaine (4 ml, 2%) combination due to the rapid effect, effective sedation and strong analgesia (5). All ultrasound examinations were performed in all biopsies with a high-resolution Toshiba Nemio 20 ultrasound scanner (Toshi-

ba Medical Systems Co, Ltd, Tokyo, Japan) equipped with a 3.5 to 5 MHz convex probe. Renal biopsies were performed utilizing an 18-gauge, 15 mm biopsy needle with a springloaded biopsy gun (Pro-Mag, Manan Medical Product, Northbrook, IL). At least two core biopsy materials were obtained in both patient groups in prone position from the left kidney. The main characteristic of Group A was that the sampling of the renal cortex was obtained randomly at the lower pole. In Group A, local anaesthetic was introduced to infiltrate the skin, subcutaneous and perinephric tissues, primarily. A small stab wound was cutted with scalpel to simplify passage of the biopsy needle access. Then the biopsy needle was inserted towards to the lower pole of the left kidney. When the first

5/08/14 13:33

208

JBR–BTR, 2014, 97 (4)

Fig. 3. — Needle pathway.

radiologist holding the probe for real-time US guidance, the other radiologist advanced the needle to be eligible for tru-cut renal biopsy. The main characteristic of Group B is that the biopsy needle route with angle between 45-60 degrees with reference to the renal capsule or base line of the convex probe as much as possible with the tip of the needle directed away from the renal hilum for receiving into the corticomedullary region (Fig. 1, 2). This tract passed from the ‘Brödel’s line’ (a relatively avascular line between the anterior and posterior segmental branches of the renal artery) which is located posterior to the lateral convex border of the kidney (Fig. 3) (6). We also used color Doppler mode imaging to avoid major vascular structures on the needle pathway.The central echogenic hilum was avoided (Fig. 4). Postprocedural follow-up In both groups, patients were instructed to remain in bed with a sandbag at back for 24 h following the biopsy. Vital signs were measured at half hour intervals for the first two hours and hourly for a day thereafter. Each urine sample voided was examined for gross hematuria for a day. Follow-up hemoglobin measurements were performed 2–4 and 6–12 h after biopsy. Doppler US examination of the punctured kidney has been performed in all patients on the next day and the patients were discharged after 24 h if there was no complication. Patients who developed macroscopic hematuria were followed up until the bleeding stopped. Patients with perirenal hematomas were discharged but ultrasonography has been repeated at a two-week interval in outpatient clinics until hematomas resolved.

Caliskan et al.indd 208

Fig. 4. — Color Doppler mode imaging to avoid major vascular structures on the needle pathway.

Table I. — Clinical diagnosis prior to biopsy. Group A (n)

Group B (n)

Total, n (%)

Nephrotic syndrome Nephritic syndrome Hematuria and/or proteinurea ARF/CRF whose cause is unknown Macroscopic hematurea Congenital nephrotic syndrome

60 25 25 14 3 1

75 39 24 23 5 0

135 (45.9%) 64 (21.7%) 49 (16.6%) 37 (12.6%) 8 (2.7%) 1 (0.3%)

Total

128

166

294

Outcome analysis

Statistical analysis

All biopsy specimens were studied by light microscopy and immunofluorescence microscopy at the Pathology Department of our hospital. Biopsy samples were subjected to preliminary evaluation under light microscope and in regions containing glomeruli; tissue was separated for immunofluorescence examination and frozen with liquid nitrogen. Biopsy material containing at least ten glomeruli has been considered as adequate for proper diagnosis. Complications have been classified as “major” and “minor”. Major complications are those which require a clinical intervention or invasive procedure, whether transfusions of blood components, the formation of an obstruction of the urinary tract with subsequent acute renal insufficiency, septicemia, or the death. Minor complications are those which do not require a clinical intervention beyond simple observation, such as transient macrohematuria, mild lumbar pain at the biopsy location, or a small perirenal hematoma (< 5 cm) that will resorb spontaneously without the need for any intervention (1, 2, 7).

Data were analyzed using the Statistical Package for Social Sciences 16.0 for Windows (SPSS Inc., Chicago, IL). Groups were compared using Fisher’s exact test in terms of the adequacy of biopsy material and complications. The results for all items were expressed as mean ± SD, assessed within a 95% reliance and at a level of p < 0.05 significance. Results Group A (traditional biopsy group) contained 128 patients (64 male, 64 female; mean age 8.5 ± 3.8 years), and Group B (angled tangential approach group) contained 166 patients (82 male, 84 female; mean age 8.1 ± 3.0 years). Among cases indicated for biopsy, clinical presumptive diagnosis were as follows: 135 (45.9%) nephrotic syndrome, 64 (21.8%) nephritic syndrome 49 (16.7%) hematurea and/ or proteinurea, 37 (12.6%) renal failure (acute and chronic) with unknown reason, eight (2.7%) macroscopic hematuria, one congenital nephrotic syndrome (Table I). Accord-

5/08/14 13:33

REAL TIME ULTRASOUND GUIDED PEDIATRIC PERCUTANEOUS RENAL BIOPSY — CALISKAN et al

209

Table II. — Histopathological diagnosis groups after biopsy. Group A n (%)

Group B n (%)

Total n (%)

Focal segmental glomerulosclerosis Minimal change disease Henoch Schönlein nephritis Membrano-proliferative glomerulonephritis Acute post infectious nephropathy Amyloidosis Congenital nephrotic syndrome Hemolytic uremic syndrome Chronic nephropathy Hereditary nephropathy Inadequate material Normal

23 (18%) 24 (18.8%) 21 (16.4%) 14 (10.9%) 13 (10.2%) 6 (4.7%) 6 (4.7%) 5 (3.9%) 5 (3.9%) 2 (1.6%) 8 (6.3%) 1 (0.8%)

38 (22.9%) 29 (17.5%) 32 (19.3%) 13 (7.8%) 15 (9%) 9 (5.4%) 9 (5.4%) 9 (5.4%) 6 (3.6%) 2 (1.2%) 1 (0.6%) 3 (1.8%)

61 (20.7%) 53 (18%) 53 (18%) 27 (9.2%) 28 (9.5%) 15 (5.1%) 15 (5.1%) 14 (4.8%) 11 (3.7%) 4 (1.4%) 9 (3.1%) 4 (1.4%)

TOTAL

128 (100%)

166 (100%)

294 (100%)

ing to the histopathological results, 61 (20.7%) cases were diagnosed with focal segmental glomerulosclerosis, 53 (18%) with minimal change disease; 53 (18%) with Henoch Schönlein nephritis, 27 (9.2%) with membrano-proliferative glomerulonephritis, 28 (9.5%) with acute post infectious nephropathy, 15 (5.1%) (5.1%) with with amyloidosis; 15 congenital nephrotic syndrome, 14 (4.8%) with hemolytic uremic syndrome, 11 (3.7%) with chronic nephropathy, and four (1.4%) with hereditary nephropathy (Table II). Consequently, nine cases (eight cases in Group A, one case in Group B) were not established any diagnosis due to inadequate material (3.1%). Inadequate material rates difference between groups was found to be statistically significant (p = 0.006). In four cases (three cases in Group A, one case in Group B) renal biopsies revealed normal renal tissue. While a major complication (hemoperitoneum requiring transfusion) occurred in one case in Group A, no major complications were seen in Group B. The most frequently occurring minor complication was macroscopic hematuria, which occurred at the rate of 20.3% in Group A and 9.6% in Group B. Hematoma was detected in four cases in Group A and in three cases in Group B. Discussion Renal biopsy has become a fundamental diagnostic tool for the diagnostic assessment of many renal diseases in nephrology. US-guidence of renal biopsy is preferred by many practitioners due to providing much benefit such as represents

Caliskan et al.indd 209

a real-time imaging, low cost and without exposing ionising radiation for patients (1, 2). Percutaneous kidney biopsy is still a risky procedure although it is carried out under the guidance of ultrasonography. The success rate of the procedure is still not 100%. This may be attributed to the fact that kidney is a mobile organ and that the tissue obtained may not contain enough glomeruli to make a diagnosis (1-4, 7). Pathologists usually need more tissue to be sampled to make interpretations that are more reliable. In order to meet the demand of pathologists, thicker needles giving more tissue samples are in routine use. In cases with inadequate tissue samples repeat of the biopsy procedure is needed. However, repeat biopsy with a thick core biopsy needle increases the risk of complication while increasing diagnostic quality (3, 4). In May 2004, a consensus meeting between radiology, pathology and pediatric nefrology clinicians was performed in our hospital to find a better approach to decrease the rate of nondiagnostic biopsies. From this date on to May 2009, pediatric percutaneous renal biopsy was standardized for passing through Broödel’s line, which comprised the angled tangential approach. Patel et al described and performed this technique for US-guided renal transplant biopsies (1). In the present study, biopsy material was obtained randomly from the renal parenchyma in Group A, while posterolateral area, which is from lower pole of renal parenchyma, was used in Group B. The speciality feature of the defined tangential approach is that the biopsy needle route with angle between 45-60 degrees with reference

to the renal capsule or base line of convex probe as much as possible with the tip of the needle directed away from the renal hilum for recieving into the corticomedullary region. The preferred site of the renal biopsy is the inferior pole of the left kidney because the renal pelvis and large caliber vessels are relatively far from this level (7-9). The superiority of angled tangential approach technique defined in this study over tangential technique defined by Patel et al. is that it increases rates of adequacy, and accuracy of the examination of the biopsy material in diseases involving only distal tubule and loop of Henle, rather than proximal nephron. In the study by Patel et al adequacy of the biopsy material was quite improved in glomerular diseases. However, in some specific diseases (PAN, RTA etc.) which involve components of distal nephron (distal tubuli, and loop of Henle) theoretically decrease the rates of accuracy, and adequacy. Since it is possible to obtain biopsy material from both proximal and distal components of nephron, procedural sensitivity and specificity will increase in all disease states independent of the diseased location of the nephron. In the literature, nephrotic syndrome is the most common indication for renal biopsy both in children and in adult patients, accounting for approximately 42-47% of patients (10, 11). In the present study, the most frequent indication for PRB was also nephrotic syndrome (45.9%) which was followed by nephritic syndrome (21.7%) and urinary abnormalities, for example hematuria and/or proteinuria (16.6%). The most frequent histopathological group of diseases found in studies in the

5/08/14 13:33

210

literature is primary glomerulonephritis in both children and adult patients (12, 13). IgA nephropathy is the most frequent glomerulopathy in adults whereas MPGN and FSGS are the most common in children (1113). In the present study, the most frequent histopathological finding was focal segmental glomerulosclerosis (20.7%). The second most frequent group of histopathology was minimal change disease, which was shown in 53 patients. The most striking feature in the present study is that amyloidosis was found in 15 patients which makes 5.1% of all study group and which does not exceed 0.5-3% in the literature (11, 12). Biopsy is a dynamic procedure requiring caution. Experience and patient compliance are required in order to decrease complications while obtaining adequate tissue for sampling. In the traditional approach, sometimes only the collector system and tubular structures were seen, without any glomeruli, which are nondiagnostic. Histopathologically adequate tissue sampling was reported to be at the rate of 96% in literature, while in the present study the corresponding rate was 93.7% for the traditional approach group and 99.4% for the the angled tangential approach group (p = 0.006) (14). Major complications are reported in 5-10% of the percutaneous renal biopsies in the literature (8). While microscopic hematuria is expected in all cases, macroscopic hematuria develops in 6.4-34.1% of the cases in the literature. Interobserver variability and experiences may in part explain this phenomenon. Walker et al. found that; under than 1% of those with macroscopic hematuria require blood transfusion (15). Arteriovenous fistula occurs at the rate of 0.5% and may be present months later with hematuria, hypertension or heart failure. Mortality is reported at the rate of 0.1% (4). The complication rate associated with the performance of percutaneous renal biopsy has significantly decreased in recent years as a result of the technological advances applied to the procedure. The use of automatic biopsy needles with guidence of US to localize the biopsy site and to visualize the needle trace for the execution of a safe procedure (1-3,7,8). In the present study, total of minor complications occurred in 42 cases (14.3%) and major complication in 1 case (0.3%). There was statisti-

Caliskan et al.indd 210

JBR–BTR, 2014, 97 (4)

cally significant difference between groups in terms of complication rates, which were 20.3% (n = 26) for Group A and 9.6% (n = 16) for Group B (p=0.007). In Group A, the minor complication rate (including perirenal hematoma and macroscopic hematuria) was 20.3% (n = 26) which was higher compared to the angled tangential approach was reduced to 9.6% (n = 16). In Group B, the rate of microscopic hematuria was 10%. To decrease the risk of hemorrhagic complications, we used color Doppler US guiding. using 18G needles might be an explanation. for the low incidence of perirenal hematoma. In the present study, adequate material obtained revealed a significant difference between the two groups. Simple differences in practice approach for US guided renal biopsies may give rise to statistically significant difference in efficiency and safety for in pediatric patients. Each complication and repetition occurring in invasive procedures means delay in treatment for the patient and source of hopelessness and tiredness due to loss of labor and time for the physician. In this study, it was thought that risk benefit ratio should be constantly controlled in order to reduce these adverse occurrences to minimum and to enhance efficiency. The present study has several limitations. First, only the rates of inadequate material and complications were counted to compare two renal biopsy procedures. It would be better if the mean number of glomeruli per core, the length of biopsy specimen, the puncture times are also analyzed and compared. Second, the design of the study (prospective case series and comparison to retrospective controls) was not a randomized, double-blind, placebocontrolled trial. Moreover, bias is to be anticipated for a combination of two different periods for retrospective data groups. However, we hope that this study will pioneer not only further studies on this method but also development of alternative options. Conclusion Compared with the traditional technique, angled tangential approach resulted in a higher adequate material rate and lower complication rate. These findings indicate that angled tangential approach may be

considered for pediatric percutaneous renal biopsies. References 1. Patel M.D., Phillips C.J., Young S.W., Kriegshauser J.S.: US-guided renal transplant biopsy: Efficacy of a cortical tangential approach. Radiology, 2010, 256: 290-296. 2. Levart T.K., Kenig A., Ponikvar J.B., Ferluga D., Cavic M.A., Kenda R.B.: Real-time ultrasound-guided renal biopsy with a biopsy gun in children: safety and efficacy. Acta Pediatrics, 2001, 90: 1394-1397. 3. Tang S., Li J.H., Lui S.L., Chan T.M., Cheng I.K., Lai K.N.: Free hand ultrasound-guided percutaneous renal biopsy: experience from single operator. Eur J Radiol, 2002, 41: 65-69. 4. Fogo A.: Renal Pathology. In: Barratt T.M., Avner E.D., Harmon W.E. (eds.). Pediatric Nephrology, fourth edition. Lippincott Williams & Wilkins, Baltimore, 1999, p. 391-413. 5. Astuto M., Disma N., Crimi E.: Two doses of oral ketamine, given with midazolam, for premedication in children. Minerva Anestetiol, 2002, 68: 593-598. 6. Sampaio F., Uflacker R.: Renal anatomy applied to urology, endourology, and interventional radiology. Thieme Medical Publishers, Inc., New York, 1993. 7. Upport R.N., Harisinghani M.G., GervaisD.A.: Imaging-Guided Percutaneous Renal Biopsy: Rationale and Approach. AJR, 2010, 194: 1443-1449. 8. Hussain F., Watson A.R., Hayes J., Evans J.: Standarts for renal biopsies: comparison of inpatients and day care procedures. Pediatr Nephrol, 2003,18: 53-56. 9. Whittier W.L., Korbet S.M.: Timing of complications in percutaneous renal biopsy. J Am Soc Nephrol, 2004, 15: 142-147. 10. Sumboonnanonda A., Srajai K., Vongjirad A., Suntornpoch V., Parichatikanond P.: Percutaneous renal biopsy in children. J Med Assoc Thai, 2002, 85 (Suppl. 2): S755-761. 11. Carvalho E., Faria Md.S., Nunes J.P.L., Sampaio S., Valbuena C.: Renal diseases: a 27-year renal biopsy study. J Nephrol, 2006, 19: 500-507. 12. Li L.S., Liu Z.H.: Epidemiologic data of renal diseases from a single unit in China: analysis based on 13,519 renal biopsies. Kidney Int, 2004, 66: 920923. 13. Chen H., Tang Z., Zeng C., et al.: Pathological demography of native patients in a nephrology center in China. Chin Med J, 2003, 116: 1377-1381. 14. Melk A.: Tool for renal tissue analysis. In: Geary D.F., Schaefer F. (eds.). Comprehensive pediatric nephrology, Mosby Elsevier Philadelpfia, 2008, 5561. 15. Walker P.D.: Practice guidelenes for the renal biopsy. Arch Pathol Lab Med, 2009, 133: 181-188.

5/08/14 13:33

JBR–BTR, 2014, 97: 211-216.

DIFFUSION-WEIGHTED MR IMAGING: ROLE IN THE DIFFERENTIAL DIAGNOSIS OF BREAST LESIONS C. Altay1, P. Balci1, S. Altay2, S. Karasu2, S. Saydam3, T. Canda4, O. Dicle1 Purpose: To evaluate the diagnostic value of magnetic resonance diffusion-weighted imaging (DWI) using apparent diffusion coefficient (ADC) values to the characterization of breast lesions and differentiation of benign and malignant lesions. Materials and methods: Thirty-seven women (mean age, 38 years) with 37 enrolled in the study. DWI and ADC maps in the axial plane were obtained using a 1.5 Tesla MRI device. Mean ADC measurements were calculated among cysts, normal fibroglandular tissue, benign lesions and malignant lesions were evaluated. Results: Out of 37 women, 4 had normally breast MRI findings. The diagnosis of remaining 33 patients with 37 breast lesions were as follows; malign lesions (n = 23), benign lesions (n = 10) and simple breast cyst (n = 4). The ADC values were as follows ( in units of 10-3 mm2/s ): Normal fibroglandular tissue (range: 1.39-2.06; mean:1.61 ± 0.23), benign breast lesions (range: 1.09-1.76; mean: 1,47 ± 0.25), cyts (range: 2.27-2.46, mean: 2,37 ± 0.07) and malignant breast lesions (range: 0.78-1.26, mean: 0.96 ± 0.25). The mean ADC obtained from malignant breast lesions was statistically different from that observed in benign solid lesions (p < < 0.01) and normal fibroglandular breast tissue (p < 0.01). Furthermore, the mean ADC values of benign breast lesions was not statistically different from cyst (p ≥ 0,01) and normal fibroglandular breast tissue (p ≥ 0,01). A ADC value of 1.1 × 10−3 mm2/s as a treshold value provided differantiation for malign and benign lesions, with a sensitivity of 91.3% and a specificity of 85.7% compared with conventional breast MRI values. Conclusion: DWI with quantitative ADC measurements is a reliable tool for differentiation of benign and malignant breast lesions. Key-word: Breast neoplasms, MR.

Currently mammography and ultrasonography still represents the primary imaging modalities used for breast cancer screening and diagnosis (1). Breast magnetic resonance imaging (MRI) is the problem solving method for the diagnosis of breast cancer (2). The conventional dynamic breast MRI has shown diagnostic sensitivities of 94–99% for invasive breast cancer, whereas various specificities have been reported (37– 86%) (3, 4). Diffusion-weighted imaging (DWI) is a useful method for the detection, diagnosis and staging of breast cancer. To increase the specificity of breast MRI, DWI could contribute to the correct diagnose of breast lesions. DWI is a technique that acquires an image during a single breath-hold and does not require contrast agent (5). DWI provides potentially exclusive information on the viability and cellularity of the in vivo tissue. It provides image contrast that is dependent on the molecular motion of water, which may be substantially altered by disease and tissue. Over

time, several studies on breast and abdominal organs examined with DWI were published (6-12). In these studies it was shown that apparent diffusion coefficient (ADC) values of normal tissues and lesions can be measured using diffusion-weighted images, and the differences in ADC values can be used in the differential diagnosis. The major aim of the current study was to measure the ADC values of benign and malignant breast lesions using DWI and to determine their contribution to differential diagnosis. Materials and methods Patients The study included conscious adult patient volunteers over 18 years of age with breast lesions that were detected by mammo graphy or ultrasonography having a diameter > 1 cm in our department. All patients gave their written informed consent prior to participating in the study, which had been ap-

From: 1. Department of Radiology, Dokuz Eylul University, Faculty of Medicine, Izmir, Turkey, 2. Department of Radiology, Izmir Atatürk Research and Training Hospital, Izmir, Turkey, 3. Department of General Surgery, Dokuz Eylul University, Faculty of Medicine, Izmir, Turkey, 4. Department of Pathology, Dokuz Eylul University, Faculty of Medicine, Izmir, Turkey. Address for correspondence: Dr C. Altay, M.D., Dokuz Eylul University, Faculty of Medicine, Department of Radiology. Mithatpas¸a Cad. Inciralti 35340 Izmir, Turkey. E-mail: cananaltay@yahoo.com

altay-.indd 211

proved by the local institutional review board. We joined in a prospective clinical trial 37 women (age range: 22–74 years; mean age: 47 years) with 37 breast lesions. Ethical approval of the study was obtained (Fig. 1, 2). MRI Conventional breast MR imaging, and DWI in a 1.5 T superconductor scanner (Intera, Gyroscan, Philips, Best, Holland) using a dedicated bilateral breast coil (four-channel breast array coil) consisting of 4 coil elements with 4 integrated preamplifiers. All patients underwent imaging in the prone position. A localizing sequence was followed by axial fast spin-echo T2-weighted imaging (repetition time (TR), 5056 ms; echo time (TE), 120 ms; echo train length, 15; section thickness, 4 mm; intersection spacing, 0,8 mm; matrix size, 256 x 256; field of view, 30 cm) with fat suppression spectral presaturation inversion recovery and fast spinecho T1-weighted imaging (TR, 550 ms; TE, 11 ms; echo train length, 15; section thickness, 4 mm; intersection spacing, 0,8 mm; matrix size, 256 x 256; field of view, 30 cm) with fat suppression (principle of selective excitation technique). This examination was followed by a dynamic study of the both of breast that consisted of serial imaging with a three-dimensional axial fast field echo T1-weighted sequence (FFE 3D; TR, 15 ms; TE, 5 ms; flip angle, 30°;

5/08/14 13:35

212

section thickness, 2 mm; intersection spacing, 0,4 mm; matrix size, 256 × 256; field of view, 30 cm; acquisition time 1 min per measurement) with fat suppression (principle of selective excitation technique). After the first acquisition, intravenous bolus injection of 0.1 mmol/kg body weight gadopentetate dimeglumine (Gd-DTPA, Magnevist, Bayer HealthCare, Leverkusen, Germany) at a flow rate of 3 ml/s by an automatic injector (Spectris, Medrad, Pittsburgh, USA) was performed, followed immediately by 20 ml of a saline solution. Thirty seconds after contrast agent injection, dynamic MR imaging was continued, using the same sequence parameters and tuning conditions, for a total of 7 contrast-enhanced measurements. The imaging timing of the dynamic series included pre-contrast, early arterial, delayed arterial, venous, and equilibrium phases of the breast. Subtraction of multiphasic contrast enhanced dynamic series was automatically acquired by the software of MR device. The software provided a new series by image-by-image subtraction of pre-contrast series from each post-contrast series of each patient. DWI studies were performed before contrast enhanced images were obtained. Diffusion-weighted sequences (TR/TE, 4200/95 ms; flip angle, 90°; slice thickness, 5 mm; field of view, 230 × 230 mm; matrix, 256 × 256; breath-holding time, 50 s) in the transverse plane were performed, applying gradients (in order to sensitize SE sequence to diffusion) to single-shot echo-planar sequences in all 3 axes (x, y, z), and 2 different b values (b = 0 s/mm2 and b = 1000 s/mm2). ADC maps were formed with these images. The measurements were made using by a circular region of interest (ROI) 10 mm in diameter in the target lesion and normal breast area on the ADC maps with reference to conventional MRI. In large-sized lesions the mean value of three different ROI measurements on the same slice was calculated. The measurements were performed from contrast enhanced solid parts on conventional breast MRI sequences and post-contrast images especially for heterogeneous lesions. In small-sized lesions the ADC value calculated with using a single ROI. Statistical analysis Pairwise comparisons for groups of more than 1 were performed using the Mann-Whitney U test with Benferroni correction for multiple

altay-.indd 212

JBR–BTR, 2014, 97 (4)

comparisons. ADC values were compared between malignant and benign masses using the Mann-Whitney U test. Statistical analysis was performed with SPSS software (Statistical Package for the Social Sciences, Version 16.0.2; SPSS, C hicago, III). A P value of less than 0.05 was considered as a statistically significant difference. Results Conventional breast MRI findings and demographics of patients The conventional breast MR images in 37 patients reviewed by using the Breast Imaging Reporting and Data System (BI-RADS) MR lexicon (13) by two radiologists (C.A. and P.B., with 5 and 13 years of breast MR imaging experience, respectively). All 37 lesions were included in category 4a (n = 12), category 4b (n = 17), or category 5 (n = 8) with conventional breast MRI findings. The histopathologic diagnosis was obtained for all 37 lesions The benign breast lesions included fibroadenomas (4/37, 10.8%), hamartoma (1/37, 2.7%), cysts (4/37, 10.8%), granulation tissue without underlying lesion in the region prior to surgery (2/37, 5.4%), sclerosing adenosis (1/37, 2.7%), ductal ectasia (1/37, 2.7%), and early stage fat necrosis (1/37, 2.7%). The malignant breast lesions were invasive ductal carcinomas (9/37, 24.3%), invasive lobular carcinomas (6/37, 16.2%), mixt type invasive ductal and lobular carcinomas (4/37, 10.8%), medullary carcinoma (1/37, 2.7%), and malignant epithelial tumor (1/37, 2.7%). Final diagnosis included 4 cysts, 10 benign solid lesions and 23 malignant lesions. The mean size of cysts, benign lesions and malignant lesions and was, respectively, 1.7 cm (range, 1.6-2.3 cm), 2.9 cm (range, 1.96.4 cm), 2.9 cm (range, 1.5-13.5 cm). Diffusion weighted MR imaging findings The powerful significant difference in the median ADC value of benign breast lesions (median, 1.47 ± 0.25 × 10−3 mm2/s; range, 1.09– 1.76 × 10−3 mm2/s) compared with malignant breast masses (0.96 ± 0.25 × 10−3 mm2/s; 0.78-1.26 × 10−3 mm2/s) was obtained (p < 0.01). The median ADC values of different lesion types are shown in Table II. The median ADCs of cysts (2.37 ± 0.07 × 10-3 mm2/s; 2.27-2.46 × 10−3 mm2/s) were significantly differ-

ent from the malignant breast lesions (p = 0,006) and normal fibroglandular breast tissue (1.61 ± 0.23 × 10−3 mm2/s; 1.39–2.06 × 10−3 mm2/s) (p = 0,004). There was no statistically significant difference between benign breast lesions and normal fibroglandular breast tissue (p > 0,01). There was no difference in ADC values among benign breast lesions including fibroadenomas (1.45 ± 0.17 × 10-3 mm2/s; 1.06-1.58 × 10−3 mm2/s), cysts (2.37 ± 0.07 × 10-3 mm2/s; 2.372.46 × 10−3 mm2/s), granulation tissues (1.41 ± 0.27 × 10-3 mm2/s; 1.241.48 × 10−3 mm2/s), fat necrosis (1.39 × 10-3 mm2/s), hamartoma (1.64 × 10-3 mm2/s), sclerosing adenosis (1.76 × 10-3 mm2/s) and ductal ectasia (1.38 × 10-3 mm2/s) (Table I). The mean ADC value of each type of lesion was invasive ductal carcino ma: 0.95 × 10-3 mm2/s, DCIS: 0.98 × 10-3 mm2/s. The malignant breast masses in this study could not be differentiated using DWI, and ADC values of invasive ductal carcinomas were 0.94 × 10-3 mm2/s (0.88-0.98 × 10−3 mm2/s); invasive lobular carcinomas, 0.79 × 10-3 mm2/s (0.89-1.09 × 10−3 mm2/s); ductal carcinoma in situ, 0.98 × 10-3 mm2/s (0.87-1.26 × 10−3 mm2/s); and the single case of medullary carcinoma, 0.92 × 10-3 mm2/s and malignant epithelial tumor, 1.18 × 10-3 mm2/s (Table II). On DWI, all malignancies had ADC ≤ 1.1 × 10-3 mm2/s in our study. Using a mean ADC of > 1.1 provided a sensitivity of 91.3%, a specificity of 85.7%, and an accuracy of 89.1% to designation lesions as benign. Discussion Conventional breast MRI is the widely used diagnostic technique for evaluating the different breast disease (1). To increase the detectability of breast cancer, several techniques are used for breast MRI. Especially, dynamic-enhanced MRI provides for evaluating suspicious breast lesions and it has a very high sensitivity for (14). Aldefining breast cancer though, dynamic-enhanced breast MRI has some limitations such as it has a long relative lower specificity compared to conventional breast imaging methods (15,16). A diffusion-weighted sequence was first described by Stejskal and Tanner in 1965 (11). In the practice commonly used is an ultrafast spin echo echoplanar T2-weighted sequence. For a long time, this imaging technique has been used only for neuroradiology. Recently, increased use of DWI in practice for the

5/08/14 13:35

DWMRI OF BREAST LESIONS — ALTAY et al

Table I. — Distribution of mean ADC values in histologic types of cystic and solid benign lesions. Benign cystic and solid lesions

Mean ADC value (10-3 mm2/s)

Fibroadenoma Granulation (after surgery) Ductal ectasia Sclerosing adenosis Hamartoma Fat necrosis Cyst Total

4 2 1 1 1 1 4 14

1.45 ± 0.17 1.41 ± 0.27 1.38 1.76 1.64 1.39 2.37 ± 0.07

Table II. — Distribution of mean ADC values in histologic types of breast cancer. Malignant lesions

Mean ADC value (10-3 mm2/s)

9 6 1 1 4 21

0.94 ± 0.29 0.79 ± 0.36 0.92 1.18 0.98 ± 0.26

Invasive ductal carcinoma Solid tubular Ca Lobular Ca Medullary Ca Malign epithelial Ca Ductal carcinoma in situ Total

valuation of benign and malignant e tumors in the body such as pancreatic, uterus, hepatic, prostate and breast tumors (17, 18, 19, 20). Diffusion is the term used for the randomized microscopic movement of molecules which is known as Brownian motion and this movement is measured from mean diffusion coefficient. DWI is sensitive to this randomized movement that is measured with ADC (21). ADC mea-

surements could be affected by several factors including cellularity, permeability, capillary perfusion, temperature, magnetic sensitivity of the tissue, and motion affects the actual diffusion. DWI can be performed after strong bipolar radiofrequencies pulses are added to spin echo or gradient echo sequences, by sensitizing the molecules in tissue to diffusion. Therefore, the microscopic movement of molecules can be evaluated

213

in vivo. In vivo, the diffusion of water molecules is restricted due to macromolecules and membranes. Highly cellular tissues provides decreased to diffusion. Conversely, it increases with crashed membranes or in low cellular tissues. DWI may be evaluated quantitatively by ADC values. There are some limitations of DWI. The EPI based pulse sequence was used in the present study. EPIDWI has technical limitations such as poor spatial resolution and the potential risk of image distortion caused by post-operation clips material which results in magnetic field inhomogeneity, patient motion, and tissue-air interface. Other DWI techniques based on parallel line scan (Periodically rotated overlapping parallel lines with enhanced reconstruction- PROPELLER sequence) diffusion (18) or on the addition of parallel imaging (sensitivity encoding-SENSE sequence) diffusion (22) can help reduce distortion and may help further improve diagnostic accuracy. In this study, ADC measurements of benign and malignant breast tumors were significantly different, so that compatible with findings of previous studies (6-12). Cysts, normal fibroglandular breast tissue, and benign breast tumors had the highest ADC values, although malignant breast masses had the lowest. There are many studies of benign and malign masses discrimination of the breast. The findings of studies show that the mean ADC vlues of the malignant tumors were 1.60 ± 0.36 × 10-3 mm2/s using b value with 400 by Sinha et al. (14), 1.22 ± 0.19 × 10-3 mm2/s using b value with 700 by Kinoshita et al. (13),

Fig. 1. — A. Transverse fat saturated T1-weighted turbo s pin-echo MR image (SPIR TSE T1) from a 49-year-old female patient with a histologically proved hamartoma in the left breast (white arrows). B. The fat saturated T2-weighted image (SPIR TSE T1) also shows a slowly bright signal intensity of the lesion (arrows) with a small amount fat tissue is marked with a grey arrow in Fig. 1 A-E.

altay-.indd 213

5/08/14 13:35

214

JBR–BTR, 2014, 97 (4)

D Fig. 1. — C. The T1-weighted gradient-echo image (3D FFE) also shows a iso-intense lesion compared with normal breast tissue (white arrows). D. Diffusion-weighted image (b = 1,000 mm2/s) reveals slightly hypointensity of the mass with more clear borders (arrows). E, Apparent diffusion coefficient (ADC) was calculated. Tumor on ADC image shows isointense compared with normal parenchyma.

E 1.12 ± 0.24 × 10-3 mm2/s using b value with 750 by Woodhams et al. (17), 1.25 ± 0.29 × 10−3 mm2/s using b value with 600 by Marini et al. (16), and 0.97 ± 0.20 × 10-3 mm2/s using b value with 1000 by Guo et al. (15).

Twenty-three malignant lesions in our study had ADC values 0.92 ± 0.25 × 10−3 mm2/s (b value was chosen 1000), which is well correlated with the similar b value study results. Also, the mean ADC value of normal

breast tissues (1,61 ± 0,23 × 10-3 mm2/s) in this study was comparable with reported ADC values of 1,63 ± 0,22 ´ 10-3 mm2/s by Guo et al. (15). Guo et al. were selected the b value = 1000 so this is similar to our study. They demonstrated 93% sensitivity with the threshold 1.3 × 10−3 mm2/s mm2/s of ADC value for differentiated malignant and benign lesions (8). Although, our study showed a 91.3% sensitivity to breast cancer with a threshold of 1.1 × 10-3 mm2/s (Table III). Compared to the prior reports, our benign breast tumors had highest ADC values of, which were similar to

Table III. — Comparison of previous studies about diffusion imaging of breast lesions. “n”: number of case; *: × 10−3 mm2/s; Sens: sensitivity; Spec: specificity.

altay-.indd 214

Previous studies

«n»

«b» value

Mean ADC* value of bening tumours

Mean ADC* value of breast cysts

Mean ADC* value of malignant tumours

ADC* of normal breast tissue

Cut of value of ADC*

Sens. %

Spec. %

Kinoshita et al.

700

1.49 ± 0.18

1.21 ± 0.18

–

Sinha et al.

400

2.01 ± 0.46

2.65 ± 0.30

1.60 ± 0.36

2.37 ± 0.27

–

Guo et al.

1000

1.57 ± 0.23

2.35 ± 0.08

0.97 ± 0.20

–

1.3

Marini et al.

600

1.74 ± 0.46

2.25 ± 0.26

1.25 ± 0.29

–

Woodhams et al.

750

1.74 ± 0.46

2.25 ± 0.26

1.12 ± 0.24

2.05 ± 0.27

1.6

Current study

1000

1.74 ± 0.25

2.37 ± 0.07

0.96 ± 0.25

1.61 ± 0.23

1.1

91.3

85.7

5/08/14 13:35

DWMRI OF BREAST LESIONS — ALTAY et al

215

Fig. 2. — A. Transverse fat saturated T1-weighted turbo spin-echo MR image (SPIR TSE T1) from a 52-year-old female patient with a histologically proved invasive lobular carcinoma in the left breast (arrows). The mass shows slightly high signal intensity compared with right breast fibro glandular tissue. B. The corresponding fat saturated T2-weighted image (SPIR TSE T1) has a bright lesion within the left breast (arrows) and there is diffuse thickening and edema of the ipsilateral breast skin in Fig. 2 A-C. C. Contrast-enhanced T1-weighted 3D fast field-echo axial image (3D FFE) in early phase submitted to subtraction shows peripherally enhanced mass (arrows) and central cystic component (asterisks). D. Diffusion-weighted image (b = 1,000 mm2/s) shows a left breast mass (arrows) with solid and cystic components. Apparent diffusion coefficient (ADC) reveals restricted diffusion (arrows) in the solid component and increased diffusion (asterisks) in the cystic part of the mass.

ADC values of benign breast tumors in our study. The mean ADC values of the 4 simple cysts were 2.37 ± 0.07 × 10-3 mm2/s. These results were consistent with ADC values observed in past studies. The ADC values of the tumors were significantly correlated with tumor cellularity. Intracranial primary tumors such as gliomas and meningiomas manifested a good correlation between the ADC value and tumor cellularity (23). Low-grade tumors tend to have higher ADC values than those of high-grade tumors, which may reflect the increase of water content within the neoplastic cells or interstitial spaces. Investigators noted that tumor cellularity was inversely correlated with ADC values of tumor. In tumors, the ADC value is highest in areas of cystic necrosis,

altay-.indd 215

followed by vasogenic peritumoral edema, nonenhancing solid tumor, and enhancing solid-tumor components (24). The high cellularity typical of some benign breast lesions (intraductal papilloma and fibrocystic mastopathy) has been responsible (11-13) as the possible cause of false positive DWI findings. In our study, we had one false positive case (fibroadenoma; ADC value 1.06 × 10-3 mm2/s) using the threshold of 1.1 × 10-3 mm2/s. There are some causes affects to the ADC values in different studies. In the study which has been used low b-value DWI (in low diffusion weighting) all masses were observed as hyperintense due to T2 effect (14), whereas on high b-value studies (in high diffusion weighting) signals of masses obviously decreased due to

diffusion restriction (15,16). Although true diffusion is independent of field strength, ADC values are affected by microscopic perfusion and artifacts due to field inhomogeneity; thus, ADC values are typically lower by 2–10% at 3 T compared with values at 1.5 T (25). The choice of b values also affects the calculated ADCs, with the use of higher b values ( > 500 mm2/s) being more accurate for true diffusion and resulting in lower ADC values (17). Other methods of cancer detection in the breast include MR spectroscopy and dynamic contrast- enhanced MRI, with many of the published articles in the literature. The studies with dynamic contrastenhanced MRI have shown a variable sensitivity and specificity. In contrast to these methods, DWI has

5/08/14 13:35

216

the advantages of not requiring IV contrast material and of being simple to process. Moreover, DWI requires less time to acquire than proton spectroscopy and less technologist training to perform. Furthermore, diffusion tensor MRI used to evaluate breast tumors. Baltzer et al. were emphasized that not only mean diffusivity but also diffusion anisotropy significantly differs between different breast neoplasms (12). Our study had several limitations. First, the relatively small sample size of our study. Second limitation was the low spatial resolution due to high b value selection with using EPI sequence, especially in small breast lesions (< 1 cm). Third, ADC values were manually calculated by two radiologists. Inter- and intra-observer differences were not excluded. Finally, we did not compare the utility of conventional breast MR with DWI. Conclusion The diffusion-weighted MRI sequence is a useful diagnostic tool since it can be obtained short time, there is no need to use contrast agent. It can contribute to accurate diagnosis when discrimination of benign and malignant breast masses when use with conventional MRI sequences. DWI is likely to be particularly useful with those in whom the use of gadolinium is contraindicated. In addition, the DWI can be effective screening technique in the patients with suspicious conventional breast imaging findings. References 1. Swedish Organised Service Screening Evaluation Group. Reduction in breast cancer mortality from the organised service screening with mammography: validation with alternative analytic methods. Cancer Epidemiol Biomarkers Prev, 2006, 15: 52-56. (PMID: 16434586) 2. Warner E., Messersmith H., Causer P., Eisen A., Shumak R., Plewes D.: Systematic review: using magnetic resonance imaging to screen women at high risk for breast cancer. Ann Intern Med,, 2008, 148: 671-679. (PMID: 18458280) 3. Orel S.G., Schnall M.D.: MR imaging of the breast for the detection, diagnosis, and staging of breast cancer.

altay-.indd 216

JBR–BTR, 2014, 97 (4) Radiology, 2001, 220: 13-30. (PMID: 11425968) 4. Kuhl C.K., Mielcareck P., Klaschik S., Leutner C., Wardelmann E., Gieseke J., et al.: Dynamic breast MR imaging: are signal intensity time course data useful for differential diagnosis of enhancing lesions? Radiology, 1999, 211: 101-110. (PMID: 10189459) 5. Woodhams R., Ramadan S., Stanwell P., Sakamoto S., Hata H., et al.: Diffusion-weighted imaging of the breast: Principles and clinical applications. Radiographics, 2011, 31: 10591084. 6. Kinoshita T., Yashiro N., Ihara N., Funatu H., Fukuma E., Narita M.: D iffusion-weighted half-Fourier singleshot turbo spin echo imaging in breast tumors: differentiation of invasive ductal carcinoma from fibroadenoma. J Comput Assist Tomogr, 2002, 26: 1042-1046. (PMID: 12488758) 7. Sinha S., Lucas-Quesada FA., Sinha U., DeBruhl N., Bassett L.W.: In Vivo Diffusion-Weighted MRI of the Breast: Potential for Lesion Characterization. J Magn Reson Imaging, 2002, 15: 693-704. (PMID: 12112520) 8. Guo Y., Cai Y.Q., Cai Z.L., Gao Y.G., An N.Y., Ma L., et al.: Differentiation of clinically benign and malignant breast lesions using diffusion-weighted imaging. J Magn Reson Imaging, 2002, 16: 172-178. (PMID: 12203765) 9. Marini C., Iascconi C., Giannelli M.: Quantitative diffusion-weighted MR imaging in the differential diagnosis of breast lesion. Eur Radiol, 2007, 17: 2646-2655. (PMID: 17356840) 10. Woodhams R., Matsunaga K., Iwabuchi K., Kan S., Hata H., K uranami M., et al.: Diffusion-weighted imaging of malignant breast tumors: the usefulness of apparent diffusion coefficient (ADC) value and ADC map for the detection of malignant breast tumors and evaluation of cancer extension. J Comput Assist Tomogr, 2005, 29: 644-649. (PMID: 16163035) 11. Tozaki M., Fukuma E.: H1 MR Spectroscopy and Diffusion-Weighted Imaging of the Breast: Are They Useful Tools for Characterizing Breast Lesions Before Biopsy? AJR Am J Roentgenol, 2009, 193: 840-849. (PMID: 19696300) 12. Baltzer P.A.T., Schäfer A., Dietzel M., Grässel D., Gajda M., Camara O., et al.: Diffusion tensor magnetic resonance imaging of the breast: a pilot study. Eur Radiol, 2011, 21: 1-10. (PMID: 20668860) 13. American College of Radiology. Breast imaging reporting and data system (BI-RADS) - MRI. 1st ed. Reston, Va: American College of Radiology, 2003.

D.A., Gatsonis C.A., 14. Bluemke Chen M.H., DeAngelis G.A., DeBruhl N., Harms S., et al.: Magnetic resonance imaging of the breast prior to biopsy. JAMA, 2004, 292: 2735-2742. (PMID: 15585733) 15. Ikeda D.M., Baker D.R., Daniel B.L.: Magnetic resonance imaging of breast cancer : clinical indications and breast MRI reporting system. J Magn Reson Imaging, 2000, 12: 975-983. (PMID: 11105039) 16. Le Bihan D., Turner R., Douek P., Patronas N.: Diffusion MR imaging: clinical applications. AJR Am J Roentgenol, 1992, 159: 591-599. (PMID: 1503032) 17. Koh D.M., Collins D.J.: Diffusionweighted MRI in the body: applications and challenges in oncology. AJR Am J Roentgenol, 2007, 188: 1622-1635. (PMID: 17515386) 18. Chan I., Wells W. 3rd., Mulkern R.V., Haker S., Zhang J., Zou K.H., et al. Detection of prostate cancer by integration of line-scan diffusion, T2mapping and T2-weighted magnetic resonance imaging: a multichannel statistical classifier. Med Phys, 2003, 30: 2390-2398. (PMID: 14528961) 19. Kono K., Inoue Y., Nakayama K., et al.: The role of diffusion-weighted imaging in patients with brain tumors. AJNR Am J Neuroradiol, 2001, 22: 1081-1088. (PMID: 11415902) 20. Le Bihan D., Douek P., Argyropoulou M.: Diffusion and perfusion magnetic resonance imaging in brain tumors. Top Magn Reson Imaging, 1993, 5: 25-31. (PMID: 8416686) 21. Le Bihan D., Breton E., Lallemand D., Grenier P., Cabanis E., Laval-Jeantet M.: MR Imaging of intravoxel incoherent motions: application to diffusion and perfusion in neurologic disorders. Radiology, 1986, 161: 401-407. (PMID: 3763909) 22. Liu C., Moseley M.E., Bammer R.: Simultaneous phase correction and SENSE reconstruction for navigated multi-shot DWI with non-cartesian k-space sampling. Magn Reson Med, 2005, 54: 1412-1422. (PMID: 16276497) 23. Sugahara T., Korogi Y., Kochi M., Ikushima I., Shigematu Y., Hirai T., et al.: Usefulness of diffusion weighted MRI with echo-planar technique in the evaluation of cellularity in gliomas. J Magn Reson Imaging, 1999, 9: 53-60. (PMID: 10030650) 24. Huisman T.A., Loenneker T., Barta G., Bellemann M.E., Hennig J., Fischer J.E., et al.: Quantitative diffusion tensor MR imaging of the brain: field strength related variance of apparent diffusion coefficient (ADC) and fractional anisotropy (FA) scalars. Eur Radiol, 2006, 16: 1651-1658. (PMID: 16532356).

5/08/14 13:35

JBR–BTR, 2014, 97: 217-221.

MUCOCELE OF THE APPENDIX: CASE REPORT AND REVIEW OF LITERATURE M. Faure1, R. Salgado1,2, B. Op de Beeck1, P. Bellinck2, J.-L. Termote2, P.M. Parizel1 Mucocele of the appendix is a descriptive term of a distended, mucus-filled appendix caused by various conditions, both benign and malignant. Computed tomography is the imaging modality of choice. Correct pre-operative diagnosis is important because of the possibility of peroperative rupture and subsequent development of pseudomyxoma peritonei. It is the task of the radiologist to alert the clinician and surgeon to the presence of this entity, the potential associated complications and possible signs of malignancy. Key-word: Appendix, CT.

Case report A 48-year-old man presented with abdominal pain and a small umbilical hernia. Because of the disproportional intensity of the associated abdominal complaints with regard to the size of the hernia, a computed tomography (CT) was performed. The contrast-enhanced CT examination demonstrated a cystic, ovalshaped, thin-walled structure in the right lower quadrant (Fig. 1A) in continuity with the caecum. The wall contained punctate calcifications (Fig. 1B). There was no surrounding inflammation or abscess formation. Based on the radiological findings, an initial diagnosis of a mucocele was made. An elective appendectomy was preformed a few weeks later, during which the small umbilical hernia was also repaired. After resection, the diagnosis of a mucinous cystadenoma of the appendix was confirmed on pathological examination (Fig. 2). Review of literature Epidemiology and classification A mucocele of the appendix is a rather rare entity, seen in only 0,20,3% of appendectomy specimens (13). Initially a female predominance was reported, although this has not been confirmed in more recent literature (4). It is commonly encountered in patients between 50 and 70 years-of-age with a mean age of 55 (3, 5). Mucocele of the appendix is not a true histopathological entity. It is a macroscopical descriptive term for a distended and mucus-filled appendix of variable etiology, and is generally divided into four histological groups (6) (Table I).

The first group consists of a simple retention cyst secondary to proximal occlusion of the appendix by e.g. a fecalith or scar tissue from previous inflammation, or in rare cases due to endometriosis (24). With rising pressure, degenerative changes in the appendiceal mucosa consecutively develop. This type of mucocele is usually smaller than 2cm in diameter (3, 7). The second group, called mucosal hyperplasia, has the same features as hyperplastic colon polyps. Benign mucinous cystadenomas form the third group. Finally, the fourth group encompasses the malignant mucinous cystadenocarcinomas, characterized by glandular stromal invasion and/or tumor cells in peritoneal implants i.e. pseudomyxoma peritonei. In concordance with this classification, the course and prognosis of an appendiceal mucocele varies with the histological subtype (4-6, 8). Clinical presentation and treatment options The clinical manifestation is often non-specific, mostly presenting with

From: 1. Department of Radiology, UZ Antwerpen, Antwerp, 2. Department of Radiology, H.-Hartziekenhuis, Lier, Belgium. Address for correspondence: Dr M. Faure, Department of Radiology, UZ Antwerpen, Wilrijkstraat 10, 2650 Edegem, Belgium. E-mail: marguerite_faure@hotmail.com

faure-.indd 217

vague pain and tenderness in the right lower quadrant. An abdominal mass is sometimes palpable. Nevertheless, an appendiceal mucocele is

Fig. 1. — Sagittal (A) and axial (B) CT scan shows a cystic, oval-shaped mass in the right lower quadrant with punctuate calcification in the wall of the lesion.

14/08/14 13:10

218

JBR–BTR, 2014, 97 (4)

Table I. — Histological division of mucocele of the appendix. Simple retention cyst Mucosal hyperplasia Mucinous cystadenoma Mucinous cystadenocarcinoma

Fig. 2. — Photograph of the resected specimen.

often an incidental finding, with up to 25% of patients experiencing no related symptoms. When symptoms occur, they may be caused by complications like bowel obstruction, torsion or ureteral obstruction (3, 5, 6, 8). Since imaging studies cannot reliably differentiate between benign and malignant mucoceles, surgery with complete resection is the treatment of choice. Furthermore, a neoplastic mucocele has the potential to rupture. Standard appendectomy is sufficient for retention cysts, mucosal hyperplasia and cystadenoma. In cystadenocarcinoma, choice of treatment depends on the extent of disease. If no mesenteric or adjacent organ involvement is present, standard appendectomy with resection of the appendiceal mesentery appears to be adequate (9). A right hemicolectomy is required in complicated mucoceles which involve the terminal ileum or caecum. Imaging findings The pre-operative radiological diagnosis of an appendiceal mucocele has important clinical consequences. The surgeon must be warned of the possibility of this pathological entity, since there is always a risk of rupture during surgery with subsequent evolution to a pseudomyxoma peritonei. Second, the risk of potential malignancy must always be assessed, especially in older people. While different imaging techniques can visualize an appendiceal mucocele, it is particularly computed tomography (CT) and ultrasound which have the best diagnostic value (4, 10). Currently, CT is the modality of choice for evaluating an appendiceal

faure-.indd 218

mucocele. The typical appearance is a well-circumscribed mass with a smooth thin wall, with or without mural calcifications located in the right lower quadrant (Fig. 3) (26). However, Kim et al (4) reported that the wall can have a variable thickness with many mucoceles having a thick wall, regardless of their etiology. The density of the mass may also vary, depending on the amount of contained mucin. Mostly it has a low, cystic (water) density, although a higher soft-tissue attenuation is also possible. An important feature is the usual lack of periappendiceal inflammation or abscess (10). CT is a good technique for demonstrating the anatomic relationship between the mass and the adjacent structures. To adequately determine its anatomic localization, adequate opacification of the terminal ileum and caecum can be necessary. Finally, CT is the best available modality for detecting mural calcifications, which may be punctuate or curvilinear. The presence of these calcifications is very typical for a mucocele. Nevertheless, calcifications may be absent in up to 50% of cases, making the diagnosis less straightforward (1, 7, 11). Furthermore, the diagnosis may be further compromised in large mucoceles where it may be difficult to determine the exact anatomic relationship with the caecum (12). Some authors have suggested that ultrasound (US) can be more useful in making the diagnosis of mucocele (12, 13). US typically shows an oval-shaped cystic mass with or without acoustic shadowing, depending on the presence of mural calcifications. The internal echotexture may be variable, with internal consistency varying from water-like

18% 20% 52% 10%

to gelatinous (1, 4). This, content may be layered, causing the ‘onion skin’ sign that represents the concentric pattern of mucoid material in the lesion (11, 12, 13). Similar to CT, an important feature is the lack of inflammation, with no wall thickening (> 6 mm) as seen in acute appendicitis. The wall typically has a layered appearance with an echogenic inner layer and a hypoechogenic outer layer (4, 10). While only rarely requested, magnetic resonance imaging (MRI) can also demonstrate a cystic mass like CT or US. Given its nature, it is less suited than CT for detecting calcifications. The signal intensity of the mass depends on its content. A high fluid content will lead to a high signal intensity on T2-weighted images (T2WI), with a low signal intensity on T1-weighted images (T1WI). However, if the mass has a high mucin content it will present with a high signal on both T2WI and T1WI (7, 14). Barium enema can show indirect signs of a mucocele such as an impression on the caecum, but it can not demonstrate the mucocele itself nor its extent (1, 10). It is in practice never used for this indication. Although there is incidental mentioning of the use of virtual colonoscopy in diagnosis of mucocele, at this time there is no large scale evidence to warrant the routine use of this technique in the evaluation of mucocele (27). There are only a few references in the literature regarding the potential use of 18F-flurodeoxyglucose positron emission tomography/computerized tomography (FDG PET/CT) to detect underlying malignancy in mucoceles. These show that PET has a rather low sensitivity in detecting underlying malignancy and has limited use in the evaluation of appendiceal carcinomas (28, 29). While there are no pathognomonic signs that can differentiate between a cystadenoma and a cystadenocarcinoma, some radiological features may suggest malignancy. Solid mural wall nodules which enhance after intravenous contrast administration and presence of internal papillary vegetations are suspected signs for a

14/08/14 13:10

MUCOCELE OF THE APPENDIX — FAURE et al

219

A A

B B Fig. 3. — Coronal (A) and axial (B) CT scan shows the typical appearance of an appendiceal mucocele with cystic content and mural calcifications.

mucinous cystadenocarcinoma (4, 15, 16). Furthermore, mesenterial infiltration, peritoneal implants with or without omental cakes, and ascites are also signs that suggest a malignant origin (Fig. 4). Myxoglobulosis is a rare variant of mucocele where the appendix is filled with translucent spheres. These spheres may calcify, which makes them visible on plain films and CT scans. It is believed that they are derivatives of granulation tissue from the wall of the mucocele that loosen, necrotize and may calcify (1, 17). Complications of mucocele It is important to recognize that complications may be the first manifestation of an appendiceal mucocele. It is an infrequent cause of bowel obstruction in adults due to an intussusception or a volvulus. This gives typical imaging findings with a cystic mass as leading point, with ssociated mesenteric and obstructive a

faure-.indd 219

C Fig. 4. — Axial CT scan (A), axial MR T2 HASTE (B) and coronal MR T1 GRE (C) with gadolinium from a 75-year-old man with a mucinous cystadenocarcinoma (white arrows) show internal septations, a papillary projection and irregular mural wall thickening (white arrowheads).

signs (Fig. 5). Furthermore, it can also present as an acute appendicitis due to torsion or cause genito-urinary symptoms as a result of ureteral obstruction or bladder compression. Mucoceles, both benign and malignant, can be complicated by a superimposed infection, which causes gas bubbles or an air-fluid level within the mass (5, 6, 7, 18, 25).

Pseudomyxoma peritonei is a complex and confusing entity. It is defined by the presence of mucine and debris in the peritoneal cavity (1, 5, 10, 15, 19, 20). First it was thought only to be present in malignant mucocele (21), but more recently two different categories were defined (1, 19). The first group refers to disseminated peritoneal adenomucinosis

14/08/14 13:10

220

JBR–BTR, 2014, 97 (4)

B Fig. 5. — Axial CT scan in a 26-year-old male with appendical mucocele (A) presenting as an intussusception, note the mesenteric vessels and fat in the lumen of the colon (B).

implants caused by a localized rupture of a benign mucocele. It has an uneventful clinical course, and is thought to occur in approximately 20% of benign mucinous cystadenomas. The second group, called peritoneal mucinous carcinomatosis, is characterized by diffuse mucinous implants on the peritoneal surfaces and mucus accumulation within the peritioneal cavity. This is caused by mucinous adenocarcinoma, with a poor prognosis and no reported (5). Recurrent 5-year survival rate mucinous ascites with intestinal obstruction is the major cause of morbidity (1). CT-scan shows scalloping of liver, spleen and mesentery, corresponding to the peritoneal implants (1, 10). Ascites is of low-attenuation or slightly higher in density than a transudative (5-20 HU) ascites due to mucinous material. Mucinous nodules may be seen and may calcify, usually in a rim-like fashion. MRI can also demonstrate pseudomyxoma peritonei. T2WI give optimal contrast

faure-.indd 220

C Fig. 6. — Axial contrast-enhanced CT scan (A) and MR GRE T1 +Gd early (B) and delayed (C) phase from a 40-year-old male with pseudomyxoma peritonei: scalloping of the liver with secondary perfusion defects and calcified implants at the falciform ligament.

differentiation with normal tissue. T1WI after intravenous contrast administration is useful for the evaluation of visceral invasion (Fig. 6). Pseudomyxoma peritonei, with similar findings, can also be caused by primary ovarian processes (15, 19). In patients with pseudomyxoma peritonei from ovarian cystadenocarcinoma, a mucinous neoplasm of the appendix is nearly always pres-

ent. Whether this are two primary processes or whether the ovarian tumour is secondary to the appendiceal one remains controversial, with some studies (3, 15) reporting concomitant presence of appendiceal adenoma-type mucocele and ovarian mucinous cystadenoma. Careful examination of the ovaries in women with appendiceal mucocele is therefore recommended.

14/08/14 13:10

MUCOCELE OF THE APPENDIX — FAURE et al

221

Table II. – Differential diagnosis. Intraperitoneal lesions ovarian cysts and tumours enteric duplication cysts mesenteric and omental cysts mesenteric hematoma or tumours abdominal abscesses

Treatment consists of surgical ebulking. Complete surgical tumor d removal combined with intraoperative heated chemotherapy during surgery, followed by postoperative intraperitoneal chemotherapy (Sugar baker technique) may improve symptom-free survival (23). Finally, while some studies mention an association of roughly 20% between colonic adenocarcinoma and appendiceal mucocele due to adenoma (8, 22)], this is not universally accepted (15). Differential diagnosis The differential diagnosis of an appendiceal mucocele is broad and includes both intra- and retroperitoneal lesions (2) (Table II). Conclusion An appendiceal mucocele is a escriptive term of a distended, d mucus-filled appendix caused by various conditions, both benign and malignant. Correct pre-operative diagnosis is important because among others the possibility of peroperative rupture and subsequent development of pseudomyxoma peritonei. It is the task of the radiologist to alert the clinician and surgeon to the presence of this entity, the potential associated complications and possible signs of malignancy. References 1. Dachman A., Lichtenstein J., Friedman A.: Mucocele oft he appendix and pseudomyxoma peritonei. AJR, 1985, 144: 923-929. 2. Horgan J.G., Chow P.P., Richter J.O., et al.: CT and sonography in the recognition of mucocele of the appendix. AJR, 1984, 143: 959-962. 3. Aho A.J., Heinonen R., Lauren P.: Benign and malignant mucocele of the appendix. Acta Chir Scand, 1973, 139: 392-400. 4. Kim S.H., Lim H.K., Lee W.J., Lim J.H., Byun J.Y.: Mucocele of the appendix:

faure-.indd 221

Retroperitoneal lesions inflammation, tumours and haemorrhages lymphocele renal cysts pancreatic pseudocysts

ultrasonographic and CT findings. Abdom Imaging, 1998, 23: 292296. 5. Landen S., Bertrand C., Maddern G.J., et al.: Appendiceal mucoceles and pseudomyxoma peritonei. Surg Gynecol Obstet, 1992, 175: 401. 6. Isaacs K.L., Warshauer D.M.: Mucocele of the appendix: computed tomographic, endoscopic, and pathologic correlation. Am J Gastroenterol, 1992, 87: 787-789. 7. Pickhardt P.F., Levy A.D., Rohrmann C.A., Kende A.I.: Primary neoplasms of the appendix: radiologic spectrum of disease with pathologic correlation. Radiographics, 2003, 23: 645-662. 8. Higa E., Rosai J., Pizzimbono C.A., et al.: Mucosal hyperplasia, mucinous cystadenoma, and mucinous cystadenocarcinoma of the appendix: a reevaluation of appendiceal mucocele. Cancer, 1973, 32: 1525-1541. 9. Soweid A.M., Clarkston W.K., Andrus C.H., Janney C.G.: Diagnosis and management of appendiceal mucoceles. Dig Dis, 1998, 16: 183-186. 10. Madwed D., Mindelzun R., Jeffrey R.B. Jr.: Mucocele of the appendix: imaging findings. Am J Roentgenol, 1992, 159: 69-72. 11. Attarde V., Patil P., et al.: Sonographic appearance of a giant appendicular mucocele. J Clin Ultrasound, 2011, 39: 290-292. 12. Francica G., Lapiccirella G., Giardiello C., et al.: Giant mucocele of the appendix: clinical and imaging findings in 3 cases. J Ultrasound Med, 2006, 25: 643-648. 13. Caspi B., Cassif E., Auslender R., et al.: The onion skin sign: a specific sonographic marker of appendiceal mucocele. J Ultrasound Med, 2004, 23: 117-121. 14. Koga H., Aoyagi K., Honda H., Fujishima M.: Appendiceal mucocele: sonographic and MR imaging findings. Am J Roentgenol, 1995, 165: 1552. 15. Zissin R., Gayer G., Kots E., Apter S., Peri M., Sharipo-Feinberg M.: Imaging of mucocele of the appendix with emphasis on the CT findings: a report of 10 cases. Clin Radiol, 1999, 54: 826832. 16. Lim H.K., Lee W.J., Kim S.H., Kim B., Cho J.M., Byun J.Y.: Primary mucinous cystadenocarcinoma of the appendix:

CT findings. Am J Roentgenol, 1999, 173: 1071-1074. 17. Gonzalez J.E., Hann S.E., Trujillo Y.P.: Myxoglobulosis of the appendix. Am J Surg Pathol, 1988, 12: 962-966. 18. Mourad F.H., Hussein M., Bahlawan M., et al.: Intestinal obstruction secondary to appendiceal mucocele. Dig Dis Sci, 1999, 44: 1594-1599. 19. Ronnet B.M., Zahn C.M., Kurman R.J., Kass M.E., Sugarbaker P.H., Shmookler B.M.: Disseminated peritoneal adenomucinosis and peritoneal mucinous carcinomatosis. Am J Surg Pathol, 1995, 19: 1390-408. 20. Hinson F.L., Ambrose N.S.: Pseudomyxoma peritonei. Br J Surg, 1998, 85: 1332-1339. 21. Woodruff R., McDonald J.R.: Benign and malignant cystic tumors of the appendix. Surg Gynecol Obstet, 1940, 71: 750-755. 22. Wolff M., Ahmed N.: Epithelial neoplasms of the vermiform appendix (exclusive of carcinoid). Cancer, 1976, 37: 2511-2522. 23. Sugarbaker P.H., Jablonski K.A.: Prognostic features of 51 colorectal and 130 appendiceal cancer patients with peritoneal carcinomatosis treated by cytoreductive surgery and intraperitoneal chemotherapy. Ann Surg, 1995, 221: 124-32. 24. Tsuda M., et al.: Mucocele of the appendix due to endometriosis: A rare case report. World J Gastroenterol, 2013, 30: 5021-4. 25. Naraynsingh V., Hariharan S., S ammy I.: Cecal volvulus and mucocele of the appendix. Acta Gastroenterol Latinoam, 2010, 40: 354-6. 26. Koktener A, Akin K., Kosehan D., Dener C.: Primary appendiceal tumors: clinical imaging and pathological findings. Report of four cases. JBRBTR, 2011, 94: 63-5. 27. Lange N., Barlow D., Long J.: Mucocele of the appendix on screening CT colonography: a case report. Abdom Imaging, 2008, 33: 267-9. 28. Purandare N.C., et al.: Use of FDG/ PET CT to diagnose malignancy as the cause of mucocele of the appendix. Indian J Gastroenterol, 2013. 29. Rohani P., et al.: Use of FDG-PET Imaging for Patients with Disseminated Cancer of the Appendix. Am Surg, 2010, 76: 1338-44.

14/08/14 13:10

JBR–BTR, 2014, 97: 222-227.

Update on MRI of spondyloarthritis. Part one: the sacro-iliac joint B.C. Vande Berg, P. Omoumi, A. Larbi, F. Lecouvet, J. Malghem1

– SpA is a heterogeneous group of In recent years, there has been an chronic inflammatory rheumatic increased trend toward the use of addiseases that comprises ankylosvanced medical imaging for the diing spondylitis (AS), psoriatic agnosis and assessment of Spondyarthritis, arthritis/spondylitis assoloArthritis (SpA) (1, 2). MRI of the ciated with inflammatory bowel sacro-iliac (SI) joints has become a disease, and reactive arthritis (2). key-imaging technique for the detecAS is the disease prototype. At tion of early non-radiographic axial the other end of the spectrum, SpA and has been shown to contribUndifferentiated SpA includes paute to optimized clinical decision- tients with typical features of SpA making (3). The current paper aims at that do not fulfill the criteria of the summarizing recent advances in the other previously mentioned entiunderstanding of the disease, the ties. questions from the clinicians, as well – The pathogenic hallmark of SpA is MRI protocols and findings in SpA. inflammation at the entheses, the Pitfalls and differential diagnosis for attachment sites of ligaments, fasSI changes will be addressed.

cia or tendons onto the bone (2). Anatomic targets for axial SpA are numerous and include bony areas adjacent to joints (osteitis), capsule (capsulitis), tendons (enthesitis), and synovial tissue (syno vitis) (Fig. 1) (6). Within the SI joints, SpA involvement frequently demonstrates a patchy dis tri bution with multiple focal areas of inflammation or quiescent disease (Fig. 2). – The dissemination of changes of different age (from edema to fatty infiltration) at numerous target sites is suggestive of SpA. No singleMRI finding is specific of

What does the radiologist need to know about SpA? – Tremendous changes in the field of SpA have occurred over the last decade and have been triggered by the development of several effective therapies (4). Drug development and registration require appropriate classification criteria to better delineate the target population and drug efficacy.

Fig. 1. — Fat-saturated intermediate-weighted coronal image of the SI joints in a 34-yo man with SpA. Left SI joint synovitis: widening and increased signal intensity in the joint space (large arrow). Osteitis: extensive marrow edema-like changes in adjacent bone (arrows). Enthesitis: increased signal intensity in marrow and soft tissue adjacent to ligamentous insertion (arrowhead).

Fig. 2. — Fat-saturated intermediate-weighted (A) and T1weighted coronal (B) images in a 24-yo woman with SpA. Active inflammation of the left SI joint: edema-like marrow changes on both sides of the widened SI joint (arrowheads). Non-active involvement of the right SI joint: fat-like signal intensity marrow changes near From: 1. Medical Imaging Department, Cliniques universitaires Saint-Luc, Institut de the right SI joint. An MR study obtained recherche expérimentale et Clinique (IREC) – pôle IMAG, UCL, Brussels. 21 months earlier (not shown) had dem Address for correspondence: Pr B. Vande Berg, Medical Imaging Department, onstrated active right SI joint disease. Cliniques universitaires Saint-Luc, Institut de recherche expérimentale et Clinique (IREC) – pôle IMAG, UCL, 10 avenue Hippocrate, 1200 Brussels, Belgium.

Vande Berg et al.indd 222

5/08/14 13:44

MRI OF SPONDYLOARTHRITIS: THE SACRO-ILIAC JOINT — VANDE BERG et al

223

Fig. 3. — Transverse oblique T1-weighted image (A) of the SI joints demonstrates abnormal signal intensity in marrow adjacent to the left sacro-iliac joint suggestive of sacro-iliitis. The corresponding fat-saturated (B) also demonstrates active enthesitis in right posterior iliac wing that was not seen on the T1-weighted SE images.

Table I. — Diagnostic features for SpA (from ref 1).

SA. Distribution and patterns of lesions in the musculoskeletal system observed over time in patientswith SpA mimick those observed in the nervous system in patients with Multiple Sclerosis – Diagnostic criteria and classification systems for SpA have evolved over decades and have reached maturity. The Assessment of SpondyloArthritis international Society (ASAS) has developed and validated classification criteria that include MRI findings (Table I) (5). Back pain lasting for more than three months with

Vande Berg et al.indd 223

an age of onset before 45 years is a determinant clinical feature. Active inflammatory lesion of the SI joints at MRI or radiographic sacroiliitis is required for the fulfillment of the imaging criterion “sacro-iliitis” in the ASAS classification criteria for axial SpA. What does the rheumatologist want to know from imaging? – Imaging can provide both diagnostic as well as prognostic criteria to the clinician.

– In the setting of suggestive axial pain of more than 3 months duration in a patient with less than 45 years of age at onset of symptoms, the rheumatologist may prescribe MRI of the SI joints to look for signs of active inflammation (7). – More specific structural osseous changes are depicted in more advancedcases, and rarely occur within the two first years after the onset of the disease (8). These osseouschanges are better detected with radiography or CT than with MRI. They include

5/08/14 13:44

224

JBR–BTR, 2014, 97 (4)

Fig. 4. — Coronal oblique fat-saturated proton-density (A) and enhanced fat-saturated T1-weighted (B) images of the SI joints in a 22-yo man with SpA. Synovitis appears as abnormal signal intensity in the SI joint space (arrowheads in A) with enhanced signal after iv contrast administration (arrowheads in B).

sseous erosions, trabecular bone o sclerosis and ligament ossification with subsequent interosseous ankylosis. The presence of structural osseous changes does not implicate active disease. – Serial MR imaging may help to evaluate changes in imaging signs of inflammation in response to specific treatments. How to optimize MRI protocols in the setting of SpA? MRI acquisition protocols tailored to suspected SpA should at least maximize sensitivity for the detection of edema and include the SI joints (8). MRI of the SI joints should include one T1-weighted as well as one fluid-sensitive fat-saturated sequence (Table II). The fat-suppressed intermediate-weighted sequence has a better signal-to-noise ratio than the STIR sequence but is more susceptible to artifacts. STIR will be favored for large field-of-view images (pelvic ring imaging). Additional sequences include fat-saturated T1-weighted or gradient-echo T2*-weighted sequen ces for better detection of osseous erosions (Table II). Gadolinium-enhanced fat-saturated T1-weighted sequences enable better detection of capsulitis and synovitis (9). The use of gadoliniumenhanced sequences does not appear to consistently modify the work-up of SpA patients because erosions, capsulitis and synovitis very rarely occur, if ever, without bone marrow edema (9). Dynamic contrastenhancedMR imaging can provide

Vande Berg et al.indd 224

additional information and correlates well with clinical history, degree of inflammatory back pain, and physical examination findings in patients with acute sacro-iliitis (10).There is a correlation between the degree of uptake detected at MR imaging and the inflammatory cellularity in patients with SpA, making dynamic MR imaging helpful in monitoring pharmacologic treatment of patients with inflammatory arthropathies (11). Diffusion-weighted MR imaging may also be effective in quantifying inflammatory changes at involved skeletal sites (12). Areas of active disease consistently demonstrate hyperintense signal on ADC maps although inflammation may appear as hypo-, iso- or slightly hyperintense signal on diffusion-weighted images (12). The coronal oblique plane is the imaging plane of reference; the trans verse oblique plane enables better assessment of anterior soft tissues (useful for lesion characterization) and of posterior entheses (useful for lesion detection) (8). These 2 planes are angulated around a L-R axis to be

parallel or perpendicular to the longitudinal axis of the SI joint. Diagnosis of sacro-iliitis Inflammatory lesions of the SI joints – Active inflammatory lesions of the SI joints include bone marrow edema/osteitis and synovitis/capsulitis. – Bone marrow edema/osteitis must be present to diagnose active inflammatory disease (Table III). It can involve several different target areas including the subchondral bone marrow (adjacent to the joint) (Fig. 2), the posterior fibrous component of the SI joints, and the posterior aspects of the iliac wings (enthesitis) (Fig. 3). – Synovitis/capsulitis can be detected in the joint space and in its capsule . It is recognized on fatsaturated T1-weighted SE images after gadolinium intravenous injection because fat-suppressed fluid sensitive sequences cannot differentiate articular fluid from articular synovial pannus or inflamed capsule (Fig. 4) (9).

Table II. — Mandatory and optional MRI sequences for SI imaging. – T1-weighted SE coronal oblique sequence – Fat-saturated intermediate-weighted SE coronal oblique sequence. – T1-weighted and fat-suppressed intermediate-weighted SE transverse oblique sequences (optional). – Fat-saturated T1-weighted sequence or gradient-echo T2*-weighted sequence (optional) – Gadolinium enhanced fat-saturated T1-weighted sequence (optional)

5/08/14 13:44

MRI OF SPONDYLOARTHRITIS: THE SACRO-ILIAC JOINT — VANDE BERG et al

225

– Appears as bone marrow edema – occurs early in disease course – correlates with symptoms – is detected exclusively at MRI* – presents as high signal intensity on fat-saturated fluid-sensitive sequences – needs to be visible at least on 2 consecutive slices or 2 foci on same slice for a definite diagnosis in sacro-iliac joints

cannot be recognized on CT images. Although non-specific, it could represent areas of previous inflammation. – Soft tissue ossification appears as ligament/capsule ossification and joint space ossification. Joint ossification may start as small bony bridges arising from the subchondral bone plate that may unify from both sides and may become coalescent (Fig. 6).

* The potential value of FDG-Pet for the detection of bone inflammation remains to be assessed.

Types and amount required for diagnosis

Table III. — Bone Inflammation (osteitis).

Table IV. — Structural changes. – demonstrate a propensity to produce bone – include fatty deposition/erosion/ossification near entheses and ankylosis – occur late in disease course – correlate poorly with symptoms – are better seen on radiographs/CT than MRI (except for fatty deposition) – remain poorly understood but could partly represent a healed or quiescent stage of inflammation. – do not suffice for the definition of a positive MRI if without inflammatory changes

Structural lesions of the SI joints – Structural changes of the SI joints include bone sclerosis, erosion, peri-articular fatty deposition and soft tissue ossification (Table IV). – Bone sclerosis that generally predominates in subchondral area appears as dense areas on radiographs/CT images and with low signal on T1 and variable signal on fat-saturated fluid-sensitive sequences. Bone sclerosis may be associated with erosions.

– Erosion appears on CT images as area of resorption of subchondral bone plate that may become confluent and causes enlargement of joint space (Fig. 5). At MRI, it appears as high signal intensity lesions on fat-suppressed fluidsensitive sequences in the region of the subchondral bone. – Peri-articular fatty deposition appears as foci of increased signal on T1 and low signal on fatsuppressedsequences in the subchondral bone marrow (Fig. 2). It

lesions

– Bone marrow edema must be present either on 2 consecutive sections or at two different areas on one section to be certain of its existence. – The sole presence of synovitis, enthesitis and capsulitis without bone marrow edema (osteitis) is not sufficient for a positive MRI. Pitfalls Deficiency in fat saturation is the most frequent pitfall observed with MRI of the axial skeleton. Deficient fat saturation generates areas of high signal in bone marrow and or soft tissues of the most posterior aspectof the iliac wings and should not be confused with osteitis or enthesitis. Anatomic variants can occur in the SI joints. Frequently, an accessory SI joint can be located at the postero-superior portion of the joint and may develop degenerative changes. Differential diagnosis SI joint osteoarthritis (OA), the most frequent SI disorder, usually

Fig. 5. — AP radiograph of the SI joints (A) from a 28-yo man with SpA demonstrates left SI joint erosion with enlargement of the joint space (arrowheads) and subchondral bone sclerosis. The corresponding fat-saturated intermediate-weighted image (B) demonstrates more obvious changes with edema-like signal intensity changes on both sides of the joint space and high signal intensity in the joint space.

Vande Berg et al.indd 225

5/08/14 13:45

226

JBR–BTR, 2014, 97 (4)

Fig. 6. — Coronal T1-weighted SE image in a 24-yo male patient with SpA demonstrates bilateral SI joints ankylosis with fat-like signal intensity changes in the joint space (arrowheads).

Fig. 8. — A T1-weighted SE image of the SI joints shows ankylosis (arrows) with preserved joint space in a 70-yo man with Forestier’s disease. In Forestier’s disease, SI ankylosis results from ligamentous ossification.

remainslocalized in the anterior middle aspect of the joint where biomechanical stresses are predominant. Any bone and marrow changes can be observed on CT and MR images of SI osteoarthritis except synovitisin the joint. Variants of SI joint OA include osteitis condensans ilii (Fig. 7). Septic SI arthritis is a rare condition that involves immunocompromized patients, drug abusers, women after birth delivery and paraplegic patients with skin ulcers. Presence of soft tissue abscess or of significant soft tissue changes are distinctive features from SpA. Metabolic disorders such as hyperparathyroidism and gout may also rarely involve in the SI joints. Fracture of the sacral wings is a frequent condition in elderly patients and should not be confused with SpA. Ankylosis due to ligament ossificationis extremely frequent in Forestier’s disease (Fig. 8) and may start as early as in the third decade. In this situation, the joint space is usually respected unless very chronic, in contradistinction to case of quiescent inflammatory disease (Fig. 7). Conclusion

C Fig. 7. — CT image (A) demonstrates condensing osteitis of the iliac bone (osteitis condensans ilii) in a 42-yo woman without SpA with well-delimited subchondral iliac bone sclerosis and an articular vacuum phenomenon. On the corresponding SE T1-weighted image (B), very low signal intensity is visible in sclerotic areas. On the enhanced T1-weighted image (C), the lack of signal enhancement in marrow and in widened joint space suggests absence of osteitis and synovitis.

Vande Berg et al.indd 226

MR imaging has been validated by international experts associations as a major diagnostic tool for the early detection, classification and monitoring of SpA patients. MR imagingof the SI joints plays a crucial role for the pre-radiological detectionof these patients. Fat-saturated fluid-sensitive sequences are the most sensitive sequences for the detection of bone marrow edema adjacentto sacro-iliac and spine joints or entheses. The presence of foci of active and quiescent lesions

5/08/14 13:45

MRI OF SPONDYLOARTHRITIS: THE SACRO-ILIAC JOINT — VANDE BERG et al

in the SI joints is a key-distinctive feature. References 1. Rudwaleit M., van der Heijde D., Khan M.A., Braun J., Sieper J.: How to diagnose axial spondyloarthritis early. Ann Rheum Dis, 2004, 63: 535-543. 2. Braun J., Sieper J.: Ankylosing spondylitis. Lancet, 2007, 369 (9570): 13791390. 3. Ash Z., Marzo-Ortega H.: Ankylosing spondylitis – the changing role of imaging. Skel Radiol, 2012, 41: 10311034. 4. Braun J., et al.: 2010 update on the ASAS/EULAR recommendations for the management of ankylosing spondylitis. Ann Rheum Dis, 2011, 70: 896904. 5. Rudwaleit M., van der Heijde D., Landewé R., et al.: The development of Assessment of SpondyloAr thritis international Society classification criteria for axial spondyloarthritis. II.

Vande Berg et al.indd 227

Validation and final selec tion. Ann Rheum Dis, 2009, 68: 777-783. 6. Sieper J., Rudwaleit M., Baraliakos X., et al.: The Assessment of SpondyloArthritis international Society (ASAS) handbook: a guide to assess spondyloarthritis. Ann Rheum Dis, 2009, 68 (suppl 2): ii1-ii44. 7. Rudwaleit M., Jurik A.G., Hermann K.G., et al.: Defin ing active sacroiliitis on magnetic resonance imaging (MRI) for classification of axial spondyloarthritis: a consensual approach by the ASAS/OMERACT MRI group. Ann Rheum Dis, 2009, 68: 1520-1527. 8. Navallas M., Ares J., Beltrán B., Pilar Lisbona M., Maymó J., Solano A.: Sacroiliitis associated with axial spon dylo arthropathy: New concepts and latest trends. Radiographics, 2013, 33: 933-956. 9. De Hooghe, Van Den Berg R., NavarroCompan V., et al.: Magnetic resonance imaging of the sacro-iliac joints in the early detection os spondyloarthritis: no added value of gadolinium

227

compared with short tau inversion recovery sequence. Rheumatology, 2013, 52: 1220-1224. 10. Braun J., Bollow M., Eggens U., König H., Distler A., Sieper J.: Use of dynamic magnetic resonance imaging with fast imaging in the detection of early and advanced sacroiliitis in spondylarthropathy patients. Arthritis Rheum, 1994, 37: 1039-1045. 11. Bollow M., Fischer T., Reisshauer H., et al.: Quantitative analyses of sacroiliac biopsies in spondyloarthropathies: T cells and macrophages predominate in early and active sacroiliitis– cellularity correlates with the degree of enhancement detected by mag netic resonance imaging. Ann Rheum Dis, 2000, 59: 135-140. 12. Gaspersic N. Sersa I., Jevtic V., TomsicM., Praprotnik S.: Monitoring ankylosing spondylitis therapy by dynamic contrast-enhanced and diffusion-weighted magnetic resonance imaging. Skeletal Radiol, 2008, 37: 123-131.

5/08/14 13:45

JBR–BTR, 2014, 97: 228-232.

Update on MR imaging of spondyloarthritis. Part two: SPINE MR IMAGING B.C. Vande Berg, P. Omoumi, A. Larbi, F. Lecouvet, J. Malghem1

Introduction MRI of the sacro-iliac joints has become a key-imaging technique for the detection of early non-radiographic axial SpA and has been shown to contribute to optimized clinical decision-making (1, 2, 3). The current paper aims at summarizing the contribution of Spine MRI in patients with SpA, the optimized MRI protocols and findings at imaging. Pitfalls and differential diagnosis for spine changes will be addressed. Vertebral fractures and dural ectasia occasionally observed in long-standing SpA patients will be mentioned. Contributions of spine MR imaging in patients with SpA The role of MRI of the spine in patientswith known SpA is controversial because there is little evidence, if any, that a definite diag nosis of the disease can be accepted in patients with normal sacro-iliac joints. Despite this limitation, evidence of anterior or posterior spondylitis in three or more vertebral corners or evidence of fatty deposition in several vertebral corners is highly suggestive of SpA (4). Because of the possible confusion with degenerative disc disease, spine MRI alone should not be prescribed to answer the question of the presence or absence of SpA. Nowadays, the radiologists should be aware of the spectrum of changes that can be observed on MR images of the spine of patients with SpA for several reasons. First, Spine MRI can be the first imaging test obtained in a patient with unrecognized SpA and it may contribute to the recognition of the disease. Second, in case of changes of unknown significance at the SI joints, spine MRI can demonstrate additional features that increase the likelihood of SpA. Third, involvement of the small joints of the thoracic spine is highly suggestive of SpA because of the rare involvement of these joints in degenerative joint

Table I. — Mandatory MRI sequences for spine imaging in SpA patients • T1-weighted sagittal sequence. • Fat-suppressed intermediate-weighted SE or STIR sagittal sequence. • Fat-suppressed intermediate-weighted SE or STIR coronal sequence.

disease (5). Finally, MRI plays a crucial role in the accurate diagnosis of complications of long-standing SpA such as fracture in ankylosed spine and dural ectasia. Spine MR imaging protocol in patients with SpA Spine MRI protocol includes at least one T1-weighted and one fat-suppressed fluid-sensitive sequence (Table I). The sagittal plane is the imaging plane of reference and it should cover the most lateral aspects of the spine because of the high frequency of involvement of the zygapophyseal joints. The costo-vertebral joints of the thoracic spine can also be imaged on the sagittal images. A large field-of-view coronal STIR sequence of the lumbar spine and SI joints yields important information in the setting of SpA as it is sensitive for bone marrow edema and covers a large body segment that contains numerous possible target areas. It can be optimized by including the thoracic spine, which is frequently involved in SpA (mainly the zygoapophyseal and costo-vertebral joints). The cervical spine is not an easy area to screen for lesions, and will be imaged only if clinically indicated. Whole-body MRI is a research area in which extended anatomic coverage in combination with the use of sensitive sequences may contribute to better detection of disseminated inflammatory lesions (6, 7). A drawback of this technique is the

From: 1. Medical Imaging Department, Cliniques universitaires Saint-Luc, Institut de recherche expérimentale et Clinique (IREC) – pôle IMAG, UCL, Brussels. Address for correspondence: Pr B. Vande Berg, Medical Imaging Department, Cliniques universitaires Saint-Luc, Institut de recherche expérimentale et Clinique (IREC) – pôle IMAG, UCL, 10 avenue Hippocrate, B-1200 Brussels, Belgium.

Vanden Berg et al2.indd 228

limitation in the precise definition of the involved structures due to reduced spatial resolution and the high frequency of degenerative involvement of small joints of the body (8). Further research is needed before this technique can be implemented in routine clinical practice. Diagnosis of inflammatory involvement of the spine

Fig. 1. — Discitis and enthesitis in a 64yo woman with SpA. Fat-saturated inter mediate-weighted sagittal image. Discitis is demonstrated by the presence of in creased signal intensity in the disc space (large arrow) with extensive marrow edema-like changes in adjacent bone. Enthesitis appears as increased signal intensity in marrow at the insertion site of the anterior longitudinal ligament or on the spinous proces (arrowheads). Disc ankylosis (thin arrow) is barely visible as its signal can be similar to that of adjacent marrow.

5/08/14 13:51

MRI OF SPONDYLOARTHRITIS: SPINE MR IMAGING — VANDE BERG et al

229

Fig. 2. — Anterior and posterior enthesitis in a 62-yo woman with SpA. Fat-saturated intermediate-weighted image and SE T1-weighted sagittal image of the thoraco-lumbar junction. Multiple areas of edema-like changes are located near the insertion of the anterior longitudinal ligament (white arrowheads) or near the spinous process (arrow). Areas of fat-like signal intensity are also seen near entheses (black arrowheads) suggestive of less active disease.

Inflammatory lesions of the spine – Axial SpA frequently demonstrates a patchy distribution along the axial skeleton with multiple foci of inflammation in different anatomic regions. Active inflammatory lesions of the spine include spondylitis, spondylodiscitis, arthritis of synovial joints and enthesitis. – Spondylitis is often a triangular shaped area of marrow edema localized in the corners of vertebral bodies. It parallels the areas of ligament insertion and can be located at the anterior and posterior aspect of the vertebral bodies, near the insertion of the anterior or posterior longitudinal ligaments (Fig. 1) (9). – Spondylodiscitis is usually a semi circular area of marrow edema localizedat the cortical end-plate adjacent to intervertebral disc (Fig. 2). They are associated with end-plate erosions. En-miroir lesion is not systematic. The disc is usually preserved. Spondylodiscitis has low specificity since degenerative disc lesions can have a similar appearance. (10).

Vanden Berg et al2.indd 229

– Arthritis of synovial joints presents as bone marrow edema in articular surfaces and adjacent soft tissue edema, best seen on fat-suppressed coronal images (Fig.. 3). – Enthesitis of supraspinal, interspinal (Fig. 1) ligaments and ligament flava presents as edema in corresponding areas associated with marrow edema of adjacent bones. Moderate changes in adjacent muscles can also be seen. Fat-suppressed fluid sensitive sequence obtained in the coronal plane best depicts these changes. Structural lesions of the spine – Structural lesions of the spine include syndesmophytes, ankylosis and fatty deposition. – Syndesmophytes are linear ossifications that develop in spine ligaments (Fig. 4). – Joint ankylosis can involve the intervertebral and the synovial joints of the spine (Fig. 1, 4). At the discs, ankylosis is associated with disc ossification on radiograph/CT images and with variable signal patterns on MR images including

Fig. 3. — Costo-vertebral joint arthritis in a 31-yo male with SpA. Fat-saturated intermediate-weighted image of the lower thoracic spine. Multiple areas of edema-like changes are located near the costo-vertebral joints (white arrowheads).

fatty-like, edema-like, and fibrous-like changes. – Erosion near the entheses appears as disappearance of the normal prominent aspect of the vertebral end-plate and is usually associated with adjacent bone sclerosis (Fig. 5) – Fatty deposition can be seen on MRI in any areas in which edema is observed (see above). Types and amount required for diagnosis

lesions

– Evidence of anterior/posterior spon dylitis in 3 or more sites is highly suggestive of axial SpA. Each lesion must be seen on 2 or more consecutive sections. – The vast majority of spinal inflam matory lesions in SpA are seen in the bone (osteitis) in association

5/08/14 13:51

230

JBR–BTR, 2014, 97 (4)

with enthesitis or fibrous disc insertions. It is unclear whether synovitis (zygopapohyseal, costovertebral and costo-transverse joints) does occur alone without concomitant osteitis. Differential diagnosis

Artefacts due to partial saturation of fat signal must be recognized to avoid false positive results (Fig. 6). Corner-based vertebral lesions on MRI are frequently observed in the normal asymptomatic population

A Fig. 4. — Syndesmophytes in a 36-yo male with SpA. On the lateral radiograph of the lower thoracic spine (A), a thin vertical ossification or syndesmophyte (arrowhead) bridges the adjacent vertebral corners. On the corresponding fat-saturated intermediate-weighted sagittal image (B), the ossification is not detected. Edema-like changes (large white arrow) involve a vertebral corner that appears normal on the radiograph (small arrow in A)

B A

Fig. 5. — Erosions at entheses in a 33-yo woman with SpA. On the lateral radiograph of the thoracic spine (A), vertebral erosion appears as blunted anterior vertebral corner with adjacent sclerosis (arrowhead). On the corresponding fat-saturated intermediate-weighted sagittal image (B), trabecular sclerosis appears as low signal intensity areas (arrowhead). The nucleus pulposus are preserved.

Vanden Berg et al2.indd 230

Fig. 6. — Artifact due to deficient saturation of fat signal in a 38-yo woman with SpA. Sagittal fat-saturated intermediate-weighted image of the spine (A) demonstrates an area of increased signal at the cervico-thoracic junction related to an artifact (arrowhead) as demonstrated by similar changes in the posterior skin. On the corresponding STIR image (B), the abnormal signal area is no more present.

5/08/14 13:51

MRI OF SPONDYLOARTHRITIS: SPINE MR IMAGING — VANDE BERG et al

231

Fig. 7. — Degenerative L4-L5 changes in a 43 yo woman. The sagittal CT reformatv(A) demonstrates disc space narrowing, subtle subchondral bone plate sclerosis and small osteophytes. On the corresponding T2-weighted SE image (B), well-delimited edema-like changes involve the marrow adjacent to the vertebral end-plates. The adjacent soft tissues are preserved and the disk signal intensity is not very high.

and in patients with spine symptoms. In this condition, they are usually more extensive along the vertebral end-plate than the vertebral wall, they predominate in anterior corners, and they are preferentially located at the apex of the lumbar curvature (9). Their frequency increases with age. Extensive marrow involvement in the posterior vertebral corner is rare in the normal population. Vertebral end-plate based lesions are extremely frequent in the adult population without inflammatory changes. Marrow changes in association with degenerative disc disease are extremely common (Fig. 7). Intravertebral disc herniation may also be extremely difficult to differentiate from inflammatory discitis. Septic arthritis of small synovial joints of the spine can occur in unusual conditions including in immuno-compromised patients, in non-pyogenic infections including brucellosis or mycosis (Fig. 8).

Vanden Berg et al2.indd 231

In all these situations in which spine involvement is ambiguous, dedicated MR images of the SI joints may be obtained because SpA involvement of the spine rarely occurs without SI joint changes.

Axial complications observed in patients with long-standing SpA Vertebral fracture Spontaneous vertebral fracture is the most feared complication in patientswith advanced SpA or DISH (11). Compression vertebral fracture may be similar to those observed in elderly osteoporotic patients. However, some of these fractures can be transverse and develop in the ankylosed disc or the adjacent bone (Fig. 9). They involve the anterior and posterior elements of the spine and are unstable. In the absence of early diagnosis, they may lead to neuro-arthropathic like joint disorders or to spinal cord compres-

D Fig. 8. — Septic arthritis of the costo-vertebral joint. Bone marrow edema (arrowhead), adjacent soft tissue infiltration and costo-vertebral joint destruction are visible on the (A) T1-, (B) T2- and C) enhanced T1-weighted SE images. The CT image (D) better demonstrates joint destruction (arrow).

5/08/14 13:51

232

JBR–BTR, 2014, 97 (4)

Fig. 9. — Transverse fracture of the spine in a patient with extensive spinal ankylosis due to long-standing SpA. Sagittal CT r eformat and T1- and T2-weighted images. The transverse fracture (arrow) is located at proximity of the disk and also extends in the posterior arch. Important intraosseous changes are present due to fracture chronicity.

sion. They should not be confused with inflammatory disc changes. Conclusion MR imaging of the sacro-iliac joints has been validated by international experts associations as a major diagnostic tool for the early detection, classification and monitoring of SpA patients. The role of spine MRI remains to be clearly defined in patients with uncomplicated SpA. However, the radiologist must be aware of the wide spectrum of changes that can be observed in the spine of SpA patients. The dissemination of foci of active and quiescent lesions along the axial skeleton is a key-distinctive feature. Overdiagnosis of SpA at spine MR should be avoided. References 1. Rudwaleit M., van der Heijde D., Khan M.A., Braun J., Sieper J.: How to diagnose axial spondyloarthritis early. Ann Rheum Dis, 2004, 63: 535-543.

Vanden Berg et al2.indd 232

2. Braun J., Sieper J.: Ankylosing spondylitis. Lancet, 2007, 369 (9570): 13791390. 3. Ash Z., Marzo-Ortega H.: Ankylosing spondylitis – the changing role of imaging. Skel Radiol, 2012, 41: 10311034. 4. Herman K.G., Baraliakos X., Van der Heijde D.M., et al.: Descriptions of spinal MRI lesions and definition of a positive MRI of the spine in axial spondyloarthritis: a consensual approach by the ASAS/OMERACT MRI study group. Ann Rheum Dis, 2012, 71: 1278-1288. 5. Bochkova A.G., Levshakova A.V., Bunchuk N.V., Braun J.: Spinal inflammation lesions as detected by magnetic resonance imaging in patients with early ankylosing spondylitis are more often observed in posterior structures of the spine. Rheumatology, 2010, 49: 749-755. 6. Weckbach S.: Whole-body MRI for inflammatory arthritis and other multifocal rheumatoid diseases. Semin Musculoskelet Radiol, 2012, 16: 377388. 7. Althoff C.E., Sieper J., Song I.H., Haibel H., Weiß A., Diekhoff T., Rudwaleit M., Freundlich B., Hamm B., Hermann K.G.: Active inflammation

and structural change in early active axial spondyloarthritis as detected by whole-body MRI. Ann Rheum Dis, 2013, 72: 967-973. 8. Jurik A.G., Zejden A., Lambert R.G., Rufibach K., Hodler J., Maksymowych W.P., Duewell S., Kissling R.O., Weber U.: Pitfalls in MR morphology of the sterno-costo-clavicular region using whole-body MRI. Clin Radiol, 2013, 68: 785-791. 9. Chung C.B., Vande Berg B.C., Tavernier T., Cotten A., Laredo J.D., Vallee C., Malghem J.: End plate marrow changes in the asymptomatic lumbosacral spine: frequency, dis tribution and correlation with age and degenerative changes. Skel Radiol, 2004, 33: 399-404. 10. Canella C., Schau B., Ribeiro E., Sbaffi B., Marchiori E.: MRI in Seronegative Spondyloarthritis: Imaging Features and Differential Diagnosis in the Spine and Sacroiliac Joints. Am J Radiol, 2013, 200: 149-157. 11. Westerveld L.A., Verlaan J.J., Oner FC.: Spinal fractures in patients with ankylosing spinal disorders: a systematic review of the literature on treatment, neurological status and complications. Eur Spine J, 2009, 18: 145-165.

5/08/14 13:51

JBR–BTR, 2014, 97: 233-238.

Capillary telangiectasia of the brain: Imaging with various magnetic resonance techniques F. Gelal, L. Karakas¸, A. Sarsılmaz, K. Yücel, C. Dündar, M. Apaydın1 Brain capillary telangiectasia is an incidental vascular malformation found usually in pons and sometimes in extrapontine sites. Typical MRI features are enhancement on post contrast T1 weighted images and signal loss on g radient echo images. We evaluated 10 patients with various MR techniques. Susceptibility weighted imaging was superior to GRE T2 in showing decreased signal due to susceptibility effects. Diffusion weighted imaging and diffusion tensor imaging proved not useful in the diagnosis. Key-word: Telangiectasia.

Capillary teleangiectasia of the brain (BCT) is a vascular malformation consisting of dilated capillaries separated by normal intervening brain parenchyma (1). They are usually discovered incidentally on contrast-enhanced magnetic resonance imaging (MRI) as an enhancing lesion. Finding out a contrast enhancing lesion in the brain arises concern about primary or metastatic brain tumors, demyelinating disease, inflammation or subacute ischemia (1, 2). BCT usually does not cause symptoms, although few patients with BCT having symptoms have been reported (1, 3, 4). Therefore, the differentiation of BCT from serious abnormalities that require treatment is essential to prevent unnecessary follow up or even invasive interventions. BCT is usually invisible on computed tomography (CT) and occult on angiography (1). On MRI most of time the lesion is not visible on T1, T2 or FLAIR, although some hyper intensity can be seen on T2. On gradient-echo images BCT has been shown to be of low signal intensity (5-7). Low signal on T2*-weighted images has been attributed to susceptibility effects due to deoxyhemoglobin resulting from slow flow in the dilated vascular channels. Lately susceptibility weighted imaging (SWI) is being used in the diagnosis, since it has been shown to be more sensitive to susceptibility effects (2, 8-10). Diffusion weighted imaging (DWI) has also been used for the diagnosis of BCTs (1, 11, 12). We aimed to further elucidate the imaging features of BCTs using various MRI sequences and techniques including gradient echo T2, SWI,

DWI, Diffusion Tensor Imaging (DTI) and postcontrast imaging with spin echo T1 and 3D GRE T1. Materials and methods In this observational retrospective study, we screened the reports of 63.000 MRI examinations performed in our hospital from November 2009 until July 2012 using keywords in the hospital information system. We found 10 patients (6 female and male patients; age range, 174 84 years; mean age 49 years, median age 57 years) having lesions consistent with capillary telangiectasia of the brain. Follow up examinations ranging from 4 months to 30 months (mean interval of 14 months) were available in 6 patients. Contrast enhancing lesions, located most of the time in pons, usually slightly hyperintense on T2 weighted images, which did not have mass effect and did not show any interval change in size or structure were diagnosed as capillary telangiectasia. Signal loss on gradient echo T2 and/or SWI further contributed to the diagnosis. MR images were evaluated by two radiologists in consensus. Any accompanying vascular abnormalities were noted. MRI examinations were performed on a 1.5 Tesla scanner (GE Signa HDxt, General Electric Medical Systems, Milwaukee, WI) using 8 channel head coil. The protocol consisted of axial spin echo T2 and T1 weighted images, FLAIR T2 axial, post contrast T1 weighted images in three orthogonal planes. Various gadolinium containing contrast agents were administered at a concentration of 0.1 mmol/kg for contrast enhanced

From: 1. Department of Radiology, Izmir Katip Çelebi Üniversitesi Atatürk Eg˘itim ve Aras¸tırma Hastanesi, Basın Sitesi, Izmir, Turkey. Address for correspondence: Dr F. Gelal, M.D., Olimpiyat Köyü Atletizm Sok. 22/11, Balçova, Izmir 35330, Turkey. E-mail: fgelal@gmail.com

gelal-.indd 233

imaging. Susceptibility weighted imaging was performed in 8 patients with the following scanning parameters: GRE EPI; TR/TE, 5750/25 ms; flip angle, 90 degrees; matrix, 256 x 512; FOV, 280 x 280 mm; slice thickness, 2.4 mm; spacing, 0 mm. Minimum intensity projection images were generated using the scanner’s software. GRE T2 axial images were obtained in 6 patients with the parameters of: TR/TE, 840/24; flip angle, 25 degrees; matrix, 288x224; FOV, 240 x 180 mm; slice thickness, 5.5 mm; spacing, 2.0 mm. 1 mm3 3D T1 GRE images (BRAVO) were obtained in 7 patients after contrast administration and the parameters were: TR/TE/TI, 12.3/5.2/420 ms; flip angle, 17 degrees; matrix, 288 x 288; FOV, 230 x 170 mm; slice thickness, 1.0 mm; spacing, 0 mm. DWI was performed in 7 patients with the parameters of: TR/TE, 6000/98.8; matrix, 148x128; FOV, 270 x 270 mm; slice thickness, 5.5 mm; spacing, 0.5 mm; b = 0 and b = 1000 values. ADC maps were generated by the scanner’s software. DTI was performed in 6 patients using the following parameters: TR/TE; 6500/97.2; matrix, 128 x 128; FOV, 270 x 270 mm; slice thickness, 5.5 mm; spacing, 0 mm; 100 directions. Fractional anisotropy maps were generated offline and ROI measurements were performed from the lesion and symmetric normal parenchyma of pons. When the lesion was located at midline, ROI was placed midline at a lower slice where there was no lesion. ROI size was 83 mm2 in lesions that were 10 mm or larger; and 39 mm2 in lesions that were smaller. Two tailed student t test was used to compare ROI measurements from the lesion and normal parenchyma; p < 0.05 was considered statistically significant. The reason for scanning was headache in 4 patients, seizure in 1 patient, anxiety disorder in 1 patient and carcinoma of breast, bladder and

5/08/14 13:53

234

JBR–BTR, 2014, 97 (4) Fig. 1. — Capillary telangiectasia of the medial temporal lobe (Patient 6). A. SE T2 weighted image is normal. B. Postcontrast SE T1 weighted image shows a 7 mm enhancing medial temporal lobe lesion (arrow). It is isointense on T2, FLAIR and precontrast T1 weighted images. C. On postcontrast 3D GRE T1 enhancing lesion is less well seen (arrow). D. On SWI, the lesion shows marked signal loss (arrow).

C Fig. 2. — Pontine capillary telangiectasia associated with cavernous malformation (Patient 10). A. SE T2 weighted axial image shows cavernous malformation (arrow) in the ventral part of pons. Dorsal and caudal to it, slightly hyperintense capillary telangiectasia is seen (curved arrow). Capillary telangiectasia was isointense on T1 weighted image and FLAIR. B. Postcontrast SE T1 weighted image shows enhancing midline telangiectasia as well as draining vein (arrow). C. Cavernous malformation is noted by significant signal loss on SWI. D. On SWI, capillary telangiectasia is easily recognized by marked decrease in signal (arrow). E. However, on GRE T2 the lesion is less well seen due to less signal loss (arrow).

gelal-.indd 234

5/08/14 13:53

BRAIN CAPILLARY TELANGIECTASIA: MRI FINDINGS — GELAL et al

D lip in other 3 patients. None of the lesions in the patients with follow up, including those with primary malignancies, showed any interval change in size or structure over a mean follow up period of 14 months. In patient 6, a medial temporal lobe lesion was incidentally discovered during a pituitary MRI performed for hyperprolactinemia at an outside institution, and the lesion was presumed to be a tumor. All the lesions were incidental findings and irrelevant to patients’ complaints. Results Nine lesions were located in pons (4 midline, 3 right parasagittal, 2 left parasagittal). One lesion was in the amygdala of the right medial temporal lobe (Fig. 1). The lesions ranged from 4 mm to 13 mm in size with a mean of 8.3 mm. One lesion was slightly hypointense on T1 weighted image, while all others were iso intense. On T2 weighted images, 8 lesions were slightly hyperintense, 1 lesion was markedly hyperintense, while the temporal lobe lesion was isointense. On FLAIR T2 weighted images, 3 were slightly hyperintense and 7 were isointense (Table I). On spin echo T1 weighted images contrast enhancement was marked in 7 lesions and slight in 3 lesions, whereas on 3D GRE T1 weighted images enhancement was slight in 6 lesions and no enhancement was shown in 1 lesion. In 7 patients, in whom both sequences were obtained, spin echo T1 was superior to

gelal-.indd 235

E 3D GRE T1 in demonstrating contrast enhancement. On the other hand, 3D GRE T1 was superior to spin echo T1 in showing the enhancing vessel converging to capillary telangiectasia in 3 of 5 lesions. Signal loss on SWI was noted in all 8 patients who had this scan. In 5 lesions signal loss was marked and in 3 lesions it was slight. Out of 6 patients who had GRE T2 sequence, 3 showed slight, 1 showed marked signal loss while the other 2 did not show any decrease in signal. SWI was superior to GRE T2 in demonstrating the signal loss in capillary telangiectasia. Out of 7 patients who had DWI,in 4 patients the lesions were isointense on both DWI and ADC. In the other 3 patients, lesions were isointense, slightly hyperintense and markedly hypointense on DWI and slightly hyperintense on ADC. Six patients had DTI. There was no statistically significant difference between the fractional anisotropy values of the lesions compared with normal parenchyma (p = 0.07), although a trend toward decreased anisotropy in the lesions was found (Table I). One patient had multiple cavernous malformations (CM) both in the posterior fossa and supratentorial parenchyma accompanying pontine capillary telangiectasia. A CM in one patient (Fig. 2) and a developmental venous anomaly (DVA) in another patient (Fig. 3) were found to be located very close to pontine capillary telangiectasias. In one patient with

235

C Fig. 3. — Capillary telangiectasia of the pons associated with developmental venous anomaly (Patient 4). A. SE T2weighted image shows a slightly hyperintense lesion in the right lateral part of the pons (arrow). B. On GRE T2 the signal loss in the lesion is slight (arrow). C. On SWI the signal loss is marked (arrow). D. On postcontrast SE T1 weighted image, the lesion shows marked enhancement (arrow). E. In the neighboring slice the lesion is accompanied by a developmental venous anomaly.

pontine lesion, there was a remote DVA in subinsular region. Discussion Vascular malformations of the central nervous system have been classified into four types: arterio venous malformations, cavernous malformations, developmental venous anomalies (venous angiomas) and capillary telangiectasias (13). BCTs consist of localized collections of multiple thin-walled vascular channels interspersed within normal brain parenchyma. They constitute 16% to 20% of all central nervous system vascular malformations at autopsy series and are the second most common vascular malformation after DVAs (1, 2, 14). BCTs are most of the time asymptomatic and with the increased use of high resolution MR imaging, they are being more frequently discovered incidentally. In our series, all BCTs were incidental findings and were irrelevant to patients’ complaints. There have been few reports of symptomatic BCTs causing seizures, blurred vision, cranial nerve dysfunction, progressive spastic paraparesis, sensorineural hearing loss and even death (1, 3, 4). Hemorrhage associated with BCTs is rare, and when it occurs, it is believed to be due to the associated vascular malformation and only rarely due to (7). the capillary telangiectasia Sayama et al. reported that lesions larger than 1 cm were more prone to cause symptoms (1). Two of the

5/08/14 13:53

gelal-.indd 236

Location

Pons midline

Pons left parasagittal

Pons midline

Pons right parasagittal

Right amygdala

Pons midline

Pons left parasagittal

Pons right parasagittal

Pons midline

Patient

Size (mm)

iso

↓

iso

↑

↑ iso

iso slight

iso

↑

iso

slight

none

marked

none

Signal loss on GRE T2

↑

iso

↑

iso

↑

FLAIR

iso

↑

↑↑

↑

marked

slight

marked

slight

marked

slight

marked

slight

marked

slight

marked

Signal loss Enhancement on SWI on SE T1

slight

none

slight

Enhancement on 3D GRE T1

low

none

high

none

low

high

none

low

Visibility of enhancing vessel on SE T1

high

none

high

none

high

Visibility of enhancing vessel on 3D GRE T1

iso

↑

iso

↓↓↓

iso

DWI

↑

iso

↑

iso

↑

iso

ADC

0.342(0.06) 0.480(0.06)

0.428(0.07) 0.493(0.10)

0.400(0.11) 0.432(0.13)

0.386(0.11) 0.411(0.09)

0.312(0.12) 0.314(0.10)

CM perilesional 0.401(0.05) 0.415(0.06)

none

DVA perilesional

DVA right subinsular

multiple CM

none

Accompanying Fractional vascular Anisotropy anomaly (First:Lesion, Second: Normal symmetric parenchyma)

Table I. — Location, size, and MRI features of brain capillary telangiectasias as well as accompanying vascular anomalies (CM: cavernous malformation; DVA: developmental venous anomaly).

236 JBR–BTR, 2014, 97 (4)

5/08/14 13:53

BRAIN CAPILLARY TELANGIECTASIA: MRI FINDINGS — GELAL et al

atients in our study had lesions p larger than 1 cm, although they were asymptomatic. While pons is the most common location for BCTs, other locations have been reported. In one series, 8 of 33 lesions were located in frontal lobe, occipital white matter and basal ganglia (2). Supratentorial lesions arise more concern for malignancy than pons lesions, and these may even undergo surgical resection or biopsy (1, 7). The medial temporal lobe lesion in our series was presumed to be a tumor at an outside institution. On T2, FLAIR or noncontrast T1 weighted images, some BCTs may go undetected. On precontrast images, we had only one lesion that was not detected, while 61% and 25% of BCTs were undetected in other series (2, 6). BCTs always enhance on postcontrast T1 weighted images. In some patients, as part of our higher resolution MR imaging protocol, we used 3D GRE T1 as well as SE T1 after contrast injection and showed that spin echo T1 was better than 3D GRE T1 in demonstrating contrast enhancement, while 3D GRE T1 was better than spin echo T1 in showing visibility of the enhancing vessel converging to BCT. This finding is compatible with literature findings (2) and can be explained by the fact that spin echo T1 has higher contrast resolution and lower spatial resolution than 3D GRE T1. It is well known that BCTs demonstrate decreased signal on GRE images, probably owing to susceptibility effects of deoxyhemoglobin in the slow flow vascular channels. This finding has been found to be very helpful in the diagnosis (5, 6, 7, 12). Recently, susceptibility weighted imaging (SWI), known to be more sensitive to susceptibility effects than GRE imaging, has been used in the diagnosis of BCTs (2, 8, 9,10). SWI is a high spatial resolution 3D gradient echo MR technique which is very sensitive to intravascular venous (deoxygenated) blood as well as extravascular blood products (15). We found that SWI was more successful than GRE T2 in the diagnosis of BCTs. In our study, 2 lesions were isointense on GRE T2, while hypo intense on SWI. Other authors have also reported several cases of BCTs, undetected by GRE T2 and shown to be hypointense on SWI (2, 10). However, the difference in the capabilities of the two techniques in detecting BCTs, may, in part, be due to thinner sections used in SWI compared to thicker slices used in GRE T2.

gelal-.indd 237

DWI has also been used in the diagnosis of BCTs. In a recent paper, it was reported that all 18 BCTs in pons showed decreased signal on DWI and this helped to differentiate BCT from tumor, inflammation or ischemia (11). On the contrary, other authors have shown that only 10% of 105 BCTs were hypointense on DWI (1). Our findings support the latter study. Four of the 7 lesions were isointense on both DWI and ADC; while the other 3 showed slightly increased signal on ADC. DTI characteristics of BCTs have not been reported before. We measured fractional anisotropy (FA) values of 6 lesions in comparison with normal parenchyma. Although there was no statistically significant difference in FA values, there was a trend toward decreased anisotropy, probably reflecting loss of normal white matter tracts. Small size of some lesions might have decreased the reliability of FA measurements. A DTI study comparing FA values of BCTs and other brain lesions might be helpful in determining whether or not this technique can be used to make a diagnosis in individual patients. Capillary telangiectasia is considered to be an acquired lesion, rather than a primary developmental anomaly, and the frequent presence of an associated draining vein is believed to support this hypothesis (6). DVAs and CMs have been reported to coexist with capillary telangiectasias suggesting that venous restriction of capillary-venous outflow might have resulted in this vascular triad (16). These 3 malformations are also believed to exist on a continuum, so that a DVA may progress to a CM followed by the development of a capillary telangiectasia (17). Half of the lesions in our study had an associated drainage vein, while this was the case in 1/3 and 3/4 of lesions in two larger series (2, 6). A DVA in one patient and a CM in another were found adjacent to pontine capillary telangiectasias in our study, supporting the above assumption. None of the 6 patients with follow up, including the patient with perilesional DVA, showed any interval change in the size of the lesions or hemorrhage. Absence of histopathological examination can be considered as a limitation of our study, but it is often not possible and necessary to obtain tissue diagnosis, since BCTs are benign and have fairly typical MR imaging features. Also since this was a retrospective study, it was not possible to use all of the above MR

237

techniques in all patients. A prospective and larger study using all of these techniques in each patient may provide additional data. In conclusion; BCT is an incidental vascular malformation found usually in pons and sometimes in extrapontine sites. It is hardly visible on precontrast MRI, while it enhances on postcontrast T1 weighted images and shows decreased signal on GRE T2 or SWI. These MRI findings are fairly typical for the diagnosis. Early and proper diagnosis using MRI avoids unnecessary follow up or surgical interventions. SWI is superior to GRE T2 in showing decreased signal due to susceptibility effects. DWI is not useful in the diagnosis contrary to what was reported in a previous publication. Further studies may be needed to clarify the role of DTI in the diagnosis. BCT is sometimes associated with other vascular malformations; in that case, follow up may be necessary to detect possible complications. References 1. Sayama C.M., Osborn A.G., Chin S.S., Couldwell W.T.: Capillary telangiectasias: clinical,radiographic, and histopathological features. Clinical article. J Neurosurg, 2010, 113: 709-714 2. El-Koussy M., Schroth G., Gralla J., Brekenfeld C., Andres RH., Jung S., Shahin M.A., Lovblad KO., Kiefer C., Kottke R.: Susceptibility-weighted MR Imaging for diagnosis of capillary telangiectasia of the brain. AJNR Am J Neuroradiol, 2012, 33: 715-720. Epub 2011 Dec 22. 3. Huddle D.C., Chaloupka J.C., Sehgal V.: Clinically aggressive diffuse capillary telangiectasia of the brain stem: a clinical radiologic pathologic case study. AJNR Am J Neuroradiol, 1999, 20: 1674-1677. 4. Goyal M.K., Kumar G., Sahota P.K.: Reversible sensorineural hearing loss with normal brainstem auditory evoked potentials in pontine hemorrhage due to capillary telangiectasia. J Clin Neurosci, 2010, 17: 1198-1201. Epub 2010 Jun 8. 5. Lee R.R., Becher M.W., Benson M.L., Rigamonti D.: Brain capillarytelangiecasia: MR imaging appearance and clinicohistopathologic findings. Radiology, 1997, 205: 797-805. 6. Barr R.M., Dillon W.P., Wilson C.B.: Slow-flow vascular malformationsof the pons: capillary telangiectasias? AJNR Am J Neuroradiol, 1996, 17: 7178. 7. Castillo M., Morrison T., Shaw J.A., Bouldin T.W.: MR imaging and histologic features of capillary telangiectasia of the basal ganglia. AJNR Am J Neuroradiol, 2001, 22: 1553-1555. 8. Sadana H.K.A.K., Lim T.A.: Large pontine capillary telangiectasia detected

5/08/14 13:53

238 by susceptibility-weighted imaging but inconspicuous on diffusion weighted imaging. J HK Coll Radiol, 2010, 12: 190-193. 9. Pendharkar H.S., Thomas B., Gupta A.K.: Susceptibility-weighted imaging in capillary telangiectasia. Neurol India, 2010, 58: 618-619. 10. Yoshida Y., Terae S., Kudo K., Tha K.K., Imamura M., Miyasaka K.: Capillary telangiectasia of the brainstem diagnosed by susceptibility-weighted imaging. J Comput Assist Tomogr, 2006, 30: 980-982. 11. Finkenzeller T., Fellner F.A., Trenkler J., Schreyer A., Fellner C.: Capillary telangiectasias of the pons. Does

gelal-.indd 238

JBR–BTR, 2014, 97 (4) iffusion-weighted MR increase diagd nostic accuracy? Eur J Radiol, 2010, 74: e112-116. 12. Ozcan H.N., Avcu S., De Bleecker J., Lemmerling M.: MRI findings in giant pontine capillary telangiectasis associated with a developmental venous anomaly. JBR-BTR, 2011, 94: 293-294. 13. Dillon W.P.: Cryptic vascular malformations: controversies in terminology, diagnosis, pathophysiology, and treatment. AJNR AmJ Neuroradiol, 1997, 18: 1839-1846. 14. Chaloupka J.C., Huddle D.C.: Classification of vascular malformations of the central nervous system. Neuroimaging Clin N Am 1998, 8: 295-321.

15. Haacke E.M., Xu Y., Cheng Y.C., Reichenbach J.R.: Susceptibilityweighted imaging (SWI). Magn Reson Med, 2004, 52: 612. 16. Pozzati E., Marliani A.F., Zucchelli M., Foschini M.P., Dall’OlioM., Lanzino G.: The neurovascular triad: mixed cavernous, capillary,and venous malformations of the brainstem. J Neurosurg, 2007, 10: 1113-1119. 17. Abla A., Wait S.D., Uschold T., Lekovic G.P., Spetzler R.F.: Developmental venous anomaly, cavernous malfor mation and capillarytelangiectasia: spectrum of a single disease. Acta Neurochir (Wien), 2008, 150: 487489.

5/08/14 13:53

JBR–BTR, 2014, 97: 239-241.

SMALL BOWEL ANGIOEDEMA INDUCED BY ANGIOTENSIN CONVERTING ENZYME (ACE) INHIBITOR: US AND CT FINDINGS M.L. Coelho1, R. Amaral2, L. Curvo-Semedo3, F. Caseiro-Alves3 Small bowel angioedema induced by angiotensin converting enzyme (ACE) inhibitors is a rare and often-unrecognized condition that presents with transient abdominal pain, nausea and vomiting. We report a case diagnosed in a 36 year-old female. Ultrasound and CT showed segmental small bowel wall thickening and straightening associated with marked submucosal edema and ascites. Laboratory tests only revealed mild leukocytosis. The patient improved spontaneously. Key-words: Enzymes – Intestines.

Intestinal angioedema is a rare side effect of the angiotensin converting enzyme (ACE) inhibitor, used in arterial hypertension and congestive heart failure treatment. ACE inhibitor induced small bowel angioedema is a self-limited condition that presents with sudden acute or recurrent abdominal pain, nausea and vomiting. Only a few cases have been reported in literature (1-3). Case report A 36-year-old female was admitted to the emergency department with a 24-hours history of nausea, vomiting and moderate diffuse abdominal pain. She denied diarrhea, constipation or blood in stools, as well as recent seafood ingestion. During physical examination, no fever or signs of abdominal rebound were found. The lab tests only showed mild leuko cytosis 16,4 × 109/L, with slight neutrophilia (8 × 109/L). Serology and stool cultures were negative. She had a history of hypertension treated with enalapril for two years and was taking oral contraception. Abdominal ultrasound revealed straightening and circumferential mural thickening of some small bowel loops, with very hypoechoic submucosa and folds swelling (Fig. 1). Some of these loops were distended with fluid. A moderate volume of homogeneous ascites was present in all abdominal quadrants (Fig. 1). No mesenteric adenopathy nor increased echogenicity of mesenteric fat were found. During the examination the patient denied pain with probe compression. Contrast enhanced CT, performed hours later, confirmed concen4 tric wall thickening of at least four

Fig.1. — Ultrasound study. (A) Circumferential jejunal wall thickening (0,75 mm), with marked hypoechoic submucosa and swollen folds. Fluid distension. Ascites surrounds the bowel. (B) Moderate volume of anechoic ascites in the Morrison space.

Fig. 2. — Enhanced CT MPR axial oblique image: Straightening and wall thickening of jejunal loops surrounded by ascites.

From: 1. Hospital Infante D. Pedro - Centro Hospitalar do Baixo Vouga, Aveiro, Portugal, 2. Hospital Divino Espírito Santo, Ponta Delgada, Portugal, 3. Centro Hospitalar e Universitário de Coimbra, Coimbra, Portugal Address for correspondence: Dr M.L. Ortiz de Oliveira Coelho, M.D., Rua Manuel Bandeira, 183 hab 13 4150-479 Porto – Portugal. E-mail: mluisaooc@yahoo.com

Coelho et al.indd 239

continuous loops of jejunum, two of these appearing straightened (Fig. 2). These loops also showed the so-called “target-sign”, a wall stratification comprising mucosal

5/08/14 13:57

240

JBR–BTR, 2014, 97 (4)

Fig. 3. — Enhanced CT axial images: Segmental jejunal wall thickening and stratification (circle) – edematous hypoattenuating submucosa between mucosal and serosal enhancement – the “target sign”. Moderate volume of ascites in the Morrison space.

enhancement, edematous hypoattenuating submucosa and serosal enhancement (Fig. 3). The moderate volume of ascites was still present (Fig. 3). Mesenteric vessels were patent. No lymphadenopathy was identified. An episode of small bowel angioedema induced by ACE inhibitor was the final retained diagnosis. The patient improved spontaneously with only IV hydration and was discharged after 2 days. The ACE I was replaced by an angiotensin II receptor blocker (ARB). A control abdominal ultrasound performed 6 days later confirmed a total disappearance of the findings. Discussion Angioedema is characterized by episodes of increased capillary permeability and consequent edema of cutaneous tissues or mucosal tracts. ACE inactivates bradykinin, which is an activator of the nitric oxide system that produces vasodilatation and increases vascular permeability. Inhibition of this cascade in predisposed patients leads to angioedema involving face, tongue and upper respiratory tract (1, 2). The mechanism for bowel angioedema induced by ACE inhibitor is not known, although, as in the upper respiratory tract, it is usually a very transient condition with variable degree of severity (1). Any segment of small bowel can be affected, however the jejunum is the most common (1).

Coelho et al.indd 240

Small bowel angioedema associated with ACE inhibitor can occur at any adult age, but is more common in women (1, 2, 4). It usually presents with sudden acute or recurrent abdominal pain, nausea and vomiting (2), with onset reported between a few days and almost a decade after starting taking angiotensin-converting enzyme inhibitor (3). Pain can be severe, mimicking small bowel ischemia. Diarrhea and mild leukocytosis can occur, but small bowel obstruction and fever are not expected. Usually symptoms are self-limited and resolve between 24-36 hours with or without cessation of ACE inhibitor (1, 5). Consequently, the rapid onset and fading of the angioedema give rise to imaging findings with various degrees of expression (5). Common CT findings include: segmental small bowel wall concentric thickening and the “target” sign (submucosal marked hypoattenuation between the enhancing mucosa and serosa); straightening/elongation of bowel loops (produced by the nitric oxide, also seen in ultrasound); mild bowel fluid dilatation; mesenteric vessels engorgement; mesenteric edema and variable amount of ascites (1, 3, 5, 6). No vascular compromise or adenopathy should be found. Ultrasound is especially useful in slim patients as it permits assessment of bowel motility, which should be conserved or mildly decreased, and direct correlation between the imaging findings (as parietal bowel

thickening and straightening or ascites) and the patient’s symptoms (2, 5). In this case, the ascites and the markedly thickened, straightened and hypoechoic bowel loops, that were almost painless under probe compression, raised a strong diagnostic suspicion. When this condition is identified, only conservative treatment is precluded, as IV hydration and pain control (1, 2, 5). Undiagnosed ACE inhibitor induced small bowel angioedema can lead to unnecessary invasive examinations and procedures, including surgery (2, 4-6). Discontinuation of ACE inhibitor is mandatory to prevent recurrence (1, 2, 5). Differential diagnosis include hereditary and acquire small bowel angioedema, allergic reaction, infectious gastroenteritis, vasculitis and mesenteric ischemia (6, 7). Hereditary and acquired (paraneoplastic/auto-immune) small bowel angioedema relate to C1 esterase inhibitor deficiency are indistinguishable from the drug-induced one (5, 7). However, the hereditary form has a positive family history in 75% of the cases and usually presents in infancy or early adolescence (4). The acquired form of the disease is more easily misinterpreted, as it can be present before the diagnosis (7). of the predisposing condition Allergic reaction, especially after shellfish ingestion, can be ruled out by absence of history of exposition to those allergens. Infectious gastroenteritis in the absence of fever

5/08/14 13:57

SMALL BOWEL ANGIOEDEMA INDUCED BY ACE INHIBITOR — COELHO et al

and with a great volume of ascites is highly unlikely. Patients with small bowel involvement by vasculitis often show marked vascular engorgement, nonspecific lymphadenopathy and multifocal areas of bowel wall thickening, resulting from intramural edema and haemorrhage secondary to the vasculitis-induced ischemia. Helpful clinical clues may include gastrointestinal bleeding, cutaneous manifestations or a prior diagnosis of vasculitis. Contrast enhanced abdominal CT showing patent mesenteric vessels rules out the hypothesis of mesenteric ischemia. The number of patients taking ACE inhibitor is high and rising, so it is very important to Radiologists to know and recognize ACE inhibitor induced small bowel angioedema (8). Although this entity is considered a

Coelho et al.indd 241

diagnosis of exclusion, in the appropriate clinical setting, the combination of the imaging, clinical and laboratory findings should prompt the hypothesis of this diagnosis (2, 4). References 1. Scheirey C.D., Scholz F.J., Shortsleeve M.J., Katz D.S.: Angiotensin converting enzyme inhibitor induced small-bowel angioedema: clinical and imaging findings in 20 patients. AJR Am J Roentgenol, 2011, 197: 393-398. 2. Korniyenko A., Alviar C., Cordova J., Messerly F. Visceral angioedema due to angiotensin-converting enzyme inhibitor therapy. Cleve Clin J Med, 2011, 78: 297-304. 3. Vallurupalli K., Coakley K.: MDCT Features of Angiotensin’Converting Enzyme Inhibitor-Induced Visceral Angi oedema. AJR Am J Roentgenol, 2011, 196: 405-411.

241

4. Dobbels P., Van Overbeke L., Vanbeckevoort D., Hiele M.: Acute abdomen due to intestinal angioedema induced by ACE inhibitors: not so rare? Acta Gastroenterol Belg, 2009, 72: 455-457. 5. Locascio E.J., Mahler S.A., Arnold T.C.: Intestinal angioedema misdiagnosed as recurrent episodes of gastroenteritis. West J Emerg Med, 2010, 11: 391394. 6. Aggarwal A., Mehta N., Shah S.N.: Small bowel angioedema associated with angiotensin converting enzyme inhibitor use. J Gen Intern Med, 2011, 26: 446-447. 7. DeBacker A.I., et al.: CT of angioedema of small bowel. AJR Am J Roentgenol, 2001, 176: 649-652. 8. Adhikari S.P., Schneider J.I.: An unusual cause of abdominal pain and hypotension: angioedema of the bowel. J Emerg Med, 2009, 36: 23-25.

5/08/14 13:57

JBR–BTR, 2014, 97: 242-244.

Added value of Diffusion Weighted MR Imaging in the diagnosis of postpartum ovarian vein thrombosis K. De Cuyper1, M. Eyselbergs1,2, P. Bernard1, L. Clabout3, F.M. Vanhoenacker1,2,4 Postpartum ovarian vein thrombosis (POVT) is a rare cause of right fossa pain but the diagnosis should be considered in the clinical setting of persistent fever and lower quadrant tenderness in the postpartum period. Although ultrasound is the initial step in the diagnostic work-up right fossa pain, it is often limited by overlying bowel gas and an enlarged puerperal uterus. Therefore, most authors recommend Computed Tomography (CT) as the imaging technique of choice to confirm the clinical suspicion of POVT. Because Magnetic Resonance Imaging (MRI) is less readily available, it is rarely performed for this indication.However, MRI allows not only to make the diagnosis as accurate as CT, but moreover it provides 2 important advantages in compared to CT. First, it avoids ionizing radiation. Secondly, the use of diffusion weighted imaging (DWI) may obviate the administration of intravenous contrast. Key-words: Veins, ovarian – Veins, thrombosis – Veins, MR.

Case report A previously well 31-year-old woman who had given birth to a healthy baby eleven days previously, presented with a undulating fever and nausea that started immediately in the postpartal period after an uncomplicated vaginal delivery. Clinical examination revealed an elective tenderness in the right fossa without rebound tenderness. Rovsing’s sign, Murphy’s sign, Murphy’s punch sign and the psoas sign were all negative. Plain films of the abdomen were normal. Abdominal ultrasound examination showed a hypoechogenic polylobulated heterogeneous ovoid soft tissue mass within the pelvis (Fig. 1). The lesion was partially vascularized, as shown on a color Doppler ultrasound (Fig. 2) and remarkably tender at compression with the transducer. Besides a right hydroureteronephrosis, a normal enlarged postpartal uterus could be seen. Laboratory results showed an elevated CRP of 22 mg/dL (normal value < 0,5) and a mild leukocytosis of 9,9 * 10³/µL (normal value 4-10 * 10³/µL) without a left-shift. Urinalysis was normal and particularly couldn’t reveal any pyuria nor haematuria. Fibrin D-dimers however showed to be strongly increased to 3179 ng/mL (normal value 0-500 ng/mL). On MRI, the right ovarian vein was enlarged and its lumen was hyperintense on T2-weighted images (WI) (Fig. 3). The retroperitoneal fat surrounding the vein was replaced

Fig. 1. — Axial ultrasound shows a nonspecific cloverleafshaped mass in the right iliac fossa.

by an irregularly delineated hypointense mass. On DWI (b value = 800), the lumen of the vein was hyperintense, in keeping with diffusion restriction (Fig. 4A). The ADC map showed a focal intraluminal hypo intensity (Fig. 4B). On contrast enhanced T1-WI,there was absence of luminal enhancement in the right ovarian vein in keeping with thrombus formation (Fig. 5). The patient was treated with subcutaneous low molecular weight

From: 1. Department of Radiology, AZ Sint-Maarten, Mechelen-Duffel, 2. Department of Radiology, UZ Antwerpen, Edegem, 3. Department of Gynaecology, AZ SintMaarten, Mechelen-Duffel, 4. Faculty of Medicine and Health Sciences, University of Ghent, Ghent, Belgium. Address for correspondence: Prof dr F.M. Vanhoenacker, M.D., Ph.D., Department of Radiology, AZ Sint-Maarten, Rooienberg 25, 2570 Duffel, Belgium. E-mail: filip.vanhoenacker@telenet.be

de cuyper-.indd 242

heparin and broad spectrum anti biotics for 7 days. Further recovery was uneventful. The patient was discharged from the hospital and a control MRI examination one month later, showed a normal right ovarian vein. Thrombophilia screening tests at that time were within normal limits. Discussion Ovarian vein thrombosis is associated with a variety of pelvic conditions (1) – most notably, recent childbirth, but also pelvic inflammatory disease, malignancies, and pelvic surgery. The coincidence of pre disposing etiopathogenic factors for venous thrombosis such as hyper coagulability, hemodynamic c hanges (stasis and turbulence) and endothe-

5/08/14 13:59

POSTPARTUM OVARIAN VEIN THROMBOSIS — DE CUYPER et al

243

JA KLEUR

Fig. 2. — Axial color Doppler ultrasound shows the mass is partially vascularized.

B Fig. 4. — Axial diffusion-weighted image (b value = 800) (A) and corresponding ADC map (B). Figure A demonstrates a focal area of hyperintensity (arrow). Figure B shows the corresponding ADC map with a hypointense signal along the course of the right ovarian vein (arrow).

Fig. 3. — Axial T2-weighted image shows a rounded hyper intense structure (arrow) in the center of an ill-defined mass located at the ventral aspect of the right psoas muscle. This focal area of hyperintense signal corresponds to thrombus formation within the right ovarian vein.

lial injury (due to septic trombophlebitis) – also known as the Virchow triad-are particularly present in POVT. POVT has a reported incidence of 1:600-1:7000 deliveries. However, the real incidence is probably higher due to the nonspecific symptoms of puerperal fever and postpartal pain. Early diagnosis of POVT is of utmost importance, since complications as

de cuyper-.indd 243

Fig. 5. — Axial gadolinium contrast enhanced T1-WI confirms the lack of enhancement in the right ovarian vein (arrow) due to thrombus.

5/08/14 13:59

244

thrombus extension into the inferior vena cava or renal veins, sepsis and septic pulmonary embolism are all potentially life threatening. In the United States, POVT is estimated to cause 18 maternal deaths per million pregnancies.POVT involves the right ovarian vein in 80-90% of patients (2, 3). Ultrasound and color-Doppler may show a tubular anechoic to hypoechoic structure extending superiorly from the adnex and with absence of central Doppler flow (2, 4). Ultrasound – however – is limited due to overlying bowel gas. The diagnosis is most commonly made with contrast-enhanced CT, but CT and magnetic resonance imaging (MRI) are equally sensitive for detecting this disorder, and both are more sensitive than ultrasound (5). With unenhanced CT imaging, an ovarian thrombosis appears as a hyperdense mass or isodense thrombus in the lumen of an enlarged vessel. After injection of intravenous contrast, a hypodense filling defect is seen within an enlarged vein, and there may be a variable amount of venous wall enhancement. There is often perivascular edema and venous tortuosity. On MRI, the venous thrombus in the large, tortuous puerperal ovarian vein may appear as a hypointense filling defect on contrast enhanced T1-weighted images and may appear hyperintense on T2weighted images.This current report

de cuyper-.indd 244

JBR–BTR, 2014, 97 (4)

describes the potential benefit of DWI in the diagnosis of PVOT, showing intraluminal diffusion restriction in the thrombosed ovarian vein. A major advantage of MRI compared to CT is that the diagnosis can be made without the need of the application of an ionizing radiation.Moreover, the use of DWI may obviate the administration of intravenous contrast on MRI, whereas intravenous administration of a iodinated contrast medium is needed on CT to confirm thrombosis of the ovarian vein. Less than 1% contrast media is excreted into breast milk, out of which only 1% is absorbed by the infant’s gut and hence, according to ACR recommendations (6), it is safe for the mother and infant to continue breast-feeding after receiving contrast media. However, abstention from breast feeding for 24 hours after contrast administration is recommended in most institutions. Since the DWI sequence is able to make the diagnosis without the need of an intravenous contrast agent, any potential harm to the infant can be eliminated and continuous breast feeding can be allowed. Conclusion In conclusion, septic ovarian vein thrombophlebitis is a potentially life threatening emergency, which should be diagnosed without any delay.

DWI may be a promising imaging tool to confirm the clinical suspicion without using radiation and obviating intravenous administration of contrast. References 1. Chellman-Jeffers M.R., http://emedicine.medscape.com/ article/404364-overview, Updated: May 27, 2011. 2. Kubik-Huch R.A., Hebisch G., Huch R., Hilfiker P., Debatin J.F., Krestin G.P.: Role of duplex color Doppler ultrasound, computed tomography, and MR angiography in the diagnosis of septic puerperal ovarian vein thrombosis. Abdom Imaging, 1999, 24: 8591. 3. Dessole S., Capobianco G., Arru A., Demurtas P., Ambrosini G.: Postpartum ovarian vein thrombosis: an unpredictable event: two case reports and review of the literature. Arch Gyn Obst, 2003, 267: 242-246. 4. Hadas-Halpern I., Patlas M., Fisher D.: Postpartum ovarian vein thrombophlebitis: sonographic diagnosis. Abdom Imaging, 2002, 27: 93-95. 5. Twickler D.M., Setiawan A.T., Evans R.S., et al.: Imaging of puerperal septic thrombophlebitis: Prospective comparison of MR imaging, CT, and sonography. AJR Am J Roentgenol, 1997, 169: 1039-1043. 6. Namasivayam S., Kalra M.K., Torres W.E., Small W.C.:Adverse reactions to intravenous iodinated contrast media:a primer for radiologists. Emerg Radiol, 2006, 12: 210-215.

5/08/14 13:59

JBR–BTR, 2014, 97: 245-247.

ANGIOLEIOMYOMA IN THE SOLEUS MUSCLE A. Van Holsbeeck1, L. Van den Daelen2, E. Steenkiste3, B. Van Holsbeeck1 We present a case of angioleiomyoma, a benign angiomatous soft tissue tumor in a 52-year-old patient. We emphasize that small nodular tumors showing hypervascularity on MRI or ultrasound may be malignant and should be treated with tumor excision. Key-word: Soft tissues, neoplasms.

Case report A 52-year-old female patient presented to the orthopaedic department with intermittent right calf pain of 12 months duration. There was no clinical history of trauma. Examination showed a tender nodular swelling at the musculotendinous junction of the Achilles.

The patient was referred for sonography for the presumed diagnosis of Achilles tendinosis or tear. The examination, however, showed normal Achilles tendon and a small hypervascular lesion in the distal soleus muscle (Fig. 1). For characterization, therapy planning and local staging MR was performed. On T1-weighted sequence, signal inten-

sity of the lesion was mostly intermediate (similar to adjacent muscle) (Fig. 2A). On T2-weighted sequence, signal intensity was high (Fig. 2B). Strong enhancement was seen after intravenous contrast administration (Fig. 2C). Amongst other soft tissue lesions (see discussion), an angioleiomyoma in the soleus muscle was suggest-

Fig. 1. — Sonography of the Achilles tendon and calf muscles (A) showed a heterogeneous hypoechoic lesion with sharp delineation measuring 1 cm × 0.7 cm. The lesion is located in the distal soleus muscle. Color Doppler sonography (B) demonstrated hypervascularity of the lesion.

Fig. 2. — Sagittal T1-weighted MR- sequence (A) revealed a oval lesion located in the distal soleus muscle, anterior to the Achilles tendon (arrow). The lesion is homogeneous isointense to skeletal muscle. On sagittal fat-suppressed T2-weighted image (B), the lesion is heterogeneous, predominantly hyperintense to skeletal muscle, howFrom: Department of 1. Radiology, 2. Orthopaedic surgery and 3. Pathology, Stedelijk ever with some internal isointense foci. Ziekenhuis Roeselare, Belgium. Gadolinium enhanced sagittal fat- supAddress for correspondence: Dr A. Van Holsbeeck, Department of Radiology, Stedelijk pressed T1-weighted image (C) showed Ziekenhuis Roeselare, Rode Kruisstraat 20, 8800 Roesealre, Belgium. strong enhancement. E-mail: andries.vanholsbeeck@student.kuleuven.be

Van Holsbeeck et al.indd 245

5/08/14 14:03

JBR–BTR, 2014, 97 (4)

NO COLOR

246

A Fig. 3. — Histopathology. Low- power view (A) showed a relatively sharp delineated nodular proliferation of smooth muscle cells (arrows) surrounding multiple vascular channels (asterisk). The nodule is embedded in bundles of skeletal muscle cells (short arrow) (× 50). Immunohistochemical staining (B) was positive with desmine (muscle marker) (× 200).

ed, based on the sonographic and MR characteristics. Histopathological findings and immunohistological analysis with smooth muscle cell markers confirmed the diagnosis (Fig. 3). Discussion Angioleiomyoma is a smooth muscle tumor accounting for 5% of all benign soft tissue neoplasms. The lesion originates in the tunica media of the blood vessels. It can occur anywhere in the body, however most commonly it will be discovered in an extremity, more specifically in the lower leg. The tumor can be located either superficial, as in most cases, or deep in relation to the fascia. The peak incidence is between the fourth and sixth decades of life; there is a female preponderance (1, 2). Pain is a common presenting symptom, possibly due to contraction of the smooth muscle fibers resulting in local ischemia (2). Hasegawa et al. suggested that the pain may be mediated by irritation of nerves within the lesion (3). MR most often demonstrates a sharply delineated oval mass which is homogeneous and isointense to muscle on T1-weighted images. On T2-weighted and STIR images the lesion is mixed hyperintense and isointense to muscle. Further, the hyperintense areas on T2-weighted images typically show enhancement after intravenous contrast injection (4, 5). A radiologic-pathologic study suggested that the hyperintense areas may correspond

Van Holsbeeck et al.indd 246

to blood vessels. The isointense areas are likely related to fibrous tissue and intravascular thrombi. Frequently, a hypointense peripheral rim is present on both T1- and T2-weighted images, corresponding to a fibrous capsule (5). There are several other lesions with a rich vascular supply that should be included in the differential diagnosis. Soft tissue hemangioma is the most common soft tissue tumor. However, sonography usually shows a heterogeneous, irregular lesion and acoustic shadowing may be present due to phleboliths. On T1-weighted MR images a hemangioma is typically heterogenous with hyperintense areas corresponding to fat. Other angiomatous lesions that can present like this case include hemangioendothelioma and hemangiopericytoma. The benign form of both these tumors can strongly resemble angioleiomyoma. The more malignant forms of these vascular lesions show more aggressive features of infiltration of the surrounding tissue and by imaging these lesions are indistinguishable from angiosarcoma (6). In the lower extremity, one should also be aware of small tumors that can be hypervascular and despite their small size be malignant in nature including epithelioid sarcoma and synovial sarcoma. Both these tumors most often affect the extremities in young patients. Synovial sarcoma is located near a joint (especially the popliteal fossa). Despite its name, the tumor does not arise from synovium. Epithelioid sarcoma, an aggressive soft tissue sar-

coma, mostly occurs in the distal upper extremity, followed by the lower extremity. The tumor often shows an indolent course with high risk for recurrence and metastasis (7, 8). MR is not able to differentiate between the different histological subtypes of angioleiomyoma, and what’s more worrisome, a radiologist can mistakenly diagnose a malignant lesion as benign based only on MR features. The importance of MRI relates to its ability to distinguish soft tissue planes that allow for complete and safe surgical excision. In this patient, we were able to locate a fat plane and a muscle interface between the tumor and the Achilles and the flexor hallucis muscle respectively. Both MRI and ultrasound also enabled us to located sural and saphenous nerves at a safe distance from the mass. In conclusion, we reported a case of angioleiomyoma, a benign angiomatous soft tissue tumor. We emphasized that small nodular tumors that show hypervascularity on MRI or ultrasound can be malignant. Tumor excision is therefore strongly advised. References 1. Gupte C., Butt S.H., Tirabosco R., Saifuddin A.: Angioleiomyoma: magnetic resonance imaging features in ten cases. Skeletal Radiol, 2008, 37: 1003-1009. 2. Ramesh P., Annapureddy S.R., Kahn F., Sutaria P.D.: Angioleiomyoma: a clinical, pathological and radiological review. Int J Clin Pract, 2004, 58: 587591.

5/08/14 14:03

ANGIOLEIOMYOMA IN THE SOLEUS MUSCLE — VAN HOLSBEECK et al

3. Hasegawa T., Seiki K., Yang P., Hirose T., Hizawa K.: Mechanism of pain and cytoskeletal properties in angioleiomyomas: an immunohistochemical study. Pathol Int, 1994, 44: 66-72. 4. Yoo H.J., Choi J.-A., Chung .J-H., Oh J.H., Lee G.-K., Choi J.-Y., et al.: Angioleiomyoma in soft tissue of extremities: MRI findings. AJR, 2009, 192: 291-294.

Van Holsbeeck et al.indd 247

5. Hwang J.W., Ahn J.M., Kang H.S., Suh J.S., Kim S.M., Seo J.W.: Vascular leiomyoma of an extremity: MR imaging-pathology correlation. AJR, 1998, 171: 981-985. 6. Murphey M.D., Fairbairn K.J., Parman L.M., Baxter K.G., Parsa M.B., Smith W.S.: Musculoskeletal angiomatous lesions: Radiologicpathologic correlation. Radiographics, 1995, 15: 893-917.

247

7. Murphey M.D., Gibson M.S., JenningsB.T., Crespo-Rodriguez A.M., Fanburg-Smith J., Gajewski D.A.: Imaging of synovial sarcoma with radiologic-pathologic correlation. Radiographics, 2006, 26: 1543-1565. 8. Hanna S.L., Kaste S., Jenkins J.J., Hewan-Lowe K., Spence J.V., Gupta M., et al.: Epithelioid sarcoma: clinical, MR imaging and pathologic findings. Skeletal Radiol, 2002, 31: 400-412.

5/08/14 14:03

JBR–BTR, 2014, 97: 248-250.

PRIMARY UNDIFFERENTIATED EMBRYONAL SARCOMA OF THE LIVER MISDIAGNOSED AS HYDATID CYST IN A CHILD: A CASE REPORT AND REVIEW OF THE LITERATURE A.M. Halefoglu, A. Oz1 Primary undifferentiated embryonal sarcoma (UES) of the liver is a highly malignant mesenchymal origin tumor and has a peak incidence between the ages of 6 and 10 years. We hereby report a case of primary UES of the liver in a 7-year-old male patient who initially was misdiagnosed and treated as hydatid cyst of the liver. The tumor was occupying almost the entire right lobe of the liver and had a mostly cystic appearance with some solid components in it. Because hydatid disease is endemic in this region, it can often lead to misdiagnosis. The correct diagnosis was established after a biopsy and following neo-adjuvant chemotherapy the patient underwent a successful right hepatic lobectomy with complete resection of the tumor. The patient also received adjuvant chemotherapy and is currently disease - free in the present six month period. Primary UES of the liver has a predominantly solid appearance on US in contrast to its mostly cystic appearance on CT and MRI. These paradoxical imaging findings should be kept in mind in order to be able to distinguish this rare tumor from other entities, especially hydatid cyst. Thus, early diagnosis and prompt surgical resection of these tumors together with adjuvant and/or neo-adjuvant chemotherapy can provide complete remission. Key-words: Neoplasms, in infants and children – Sarcoma.

Primary undifferentiated embryonal sarcoma (UES) of the liver constitutes 9-15% of all hepatic tumors in children and is the third most common hepatic malignancy in children after hepatoblastoma and hepatocellular carcinoma (1). However, it is still a rare tumor and until late, less than 150 cases have been reported in the literature (2, 3). UES of the liver is most commonly seen arising in children with a peak incidence between 6 and 10 years but may also arise in adults (4). The typical radiological finding of the UES is a large, septated mass with combined cystic and solid portions. However, such imaging findings can be encountered with other liver diseases and among them hydatid cyst of the liver can easily mimic this entity. Besides, this disease is endemic in the Middle East region. Thus a careful approach in the differential diagnosis is n eeded for a correct diagnosis. Herein, we report a case of UES of the liver found in a 7 year-old boy that resembled a hydatid cyst on imaging studies. Case report A 7- year-old boy was admitted to our hospital complaining of abdominal pain, distention and respiratory distress. His physical examination revealed abdominal tenderness and rebound at the upper quadrants. A palpable, firm mass was also detect-

Fig. 1. — US of the liver demonstrates a large complex cysticsolid appearance mass located in the right lobe. The color doppler reveals no flow-related enhancement in the mass.

ed in the epigastric region. He had tachipneic breathing. Chest X ray and blood counts were found within normal limits. Serology for hepatitis B surface antigen, hepatitis C and hydatid disease were negative. Serum alpha-fetoprotein (AFP) and carcinoembryonic antigen (CEA) levels were also normal. The patient was referred to ultrasound (US) examination (Applio, SSA – 770; Toshiba, Tokyo, Japan). A mass having lobulated-contours and showing slight hyperechogenity compared to the liver parenchyma was detected in the right lobe of the liver. The lesion

From: 1. Department of Radiology, Sisli Etfal Training and Research Hospital, Sisli, Istanbul, Turkey. Address for correspondence: Dr A. M. Halefoglu, M.D., Birlik sok. Parksaray ap. No: 17/4, 34340 Levent, Istanbul, Turkey. E-mail: halefoglu@hotmail.com

halefoglu-.indd 248

had both cystic and solid components, also containing large cystic and necrotic areas. There was no flow or increased vascularity in the lesion (Fig. 1). Hydatid cyst was considered as an initial diagnosis. Following US examination a contrast-enhanced abdominal CT was performed (Somatom Sensation 16, Siemens Medical Systems, Erlangen, Germany). On CT, a heterogenous hypodense mass lesion measuring 14 x 11 cm in diameter showing minimal contrast enhancement was found in the right lobe of the liver. Portal vein could not be clearly demonstrated probably due to thrombosis. (Fig. 2). The patient also underwent a magnetic resonance imaging (MRI) for further evaluation using a 1.5 tesla magnet (GE, Signa, Milwaukee, Wisconsın, USA). On

5/08/14 14:05

EMBRYONAL SARCOMA OF THE LIVER — HALEFOGLU et al

249

Fig. 2. — Axial abdominal contrast- enhanced CT scan, a ypodense mass showing minimal contrast enhancement as h seen in the right lobe of the liver.

Fig. 3. — Coronal FSE T 2 weighted MR image, a huge mass occupying almost the entire right liver lobe. The mass has a predominantly cystic appearance with multiple septations.

Fig. 4. — Axial abdominal non-contrast CT scan, obtained 6 months later, following right lobectomy no evidence of recurrent or residual tumor.

Fig. 5. — Macroscopic gross specimen of the tumor shows a well-circumscribed, multilobulated mass with multiple blood containing cystic spaces.

these images, a 152 x 90 x 77 mm mass lesion containing cystic and solid components was detected occupying almost the entire right lobe of the liver. The mass was heterogeneous and hypointense on T1 weighted images and heterogeneous and hyperintense on T2 weighted images (Fig. 3) and showed contrast enhancement in solid portions following 0.1 mmol/kg gadolinium administration. The mass contained several septaes and hemorrhagic necrotic components. Based on imaging findings, a hydatid cyst of the liver was considered as initial diagnosis and the patient was placed on Albendazole treatment. However, in spite of medical treatment, the patient ‘ s complaints did not show any regression. Therefore, percutaneous drainage of the hepatic lesion was performed. When

halefoglu-.indd 249

approximately 200 cc fresh blood material was drained from the mass, a biopsy was performed. Histopathological evaluation demonstrated undifferentiated spindle-shaped and stellate cells with markedly atypical, hyperchromatic nuclei with an abundant myxoid stroma. Based on these findings, the diagnosis was established to be primary UES of the liver. The patient recevied a three cure neo-adjuvant chemotherapy before surgery. Then, a laparotomy was performed and the mass was removed with a right hepatic lobectomy (Fig. 4). The macroscopic gross specimen showed a well-circumscribed, multilobulated mass with multiple blood containing cystic spaces (Fig. 5). Following surgery, the patient continued to receive adjuvant chemotherapy treatment and a bone scintigraphy was performed

which resulted as negative. Currently at follow-up, although the patient is still in his 6 months, there is n either recurrence nor any distant metastase on imaging studies. Discussion UES is a highly malignant primitive mesenchymal tumor that occurs predominantly in children and has a predilection for the ages of 6 to 10 years. Although it represents about 9-15% of all hepatic tumors in children, only about 150 cases have been reported in the literature (2, 3). UES is found more frequently in the right lobe of the liver (59%) than the left lobe (22%), but sometimes simultaneously in bilateral lobes (20%) (5). Diagnosis of UES is always challenging due to the lack of specific

14/08/14 13:13

250

presenting symptoms, lack of serological markers, nonspecific findings on radiological imaging and the rarity of the disease. The histogenesis of this tumor still remains undetermined. Some authors have claimed that it is the malignant counterpart of mesenchymal hamartoma (2). Patients frequently present with abdominal pain or a palpable abdominal mass. Other symptoms are anorexia, nausea, vomiting, weight loss and jaundice. Due to these nonspecific symptoms UES sometimes can be misdiagnosed as acute appendicitis (6). UES often show a misleading cystic appearance on CT and MRI in contrast to a predominantly solid appearance on US (7). The typical finding of UES in US is a single, large, solid echogenic mass with a cystic portion (8). CT scan typically demonstrates a large mass with cystic attenuation, while on MRI, large portions of the mass are hypointense on T 1 weighted images and have a high signal intensity on T 2 weighted images. With gadolinium administration, there is mild heterogenous enhancement in most of the tumor, consistent with extensive central necrosis or cystic change. This cystic appearance may lead to misdiagnosis such as hydatid cyst or amebic abscess as in our case. In the literature there have been several case reports of UES of the liver being mistaken for hydatid disease (9, 10, 11). According to Buetow et al. (7) the increased water content within the abundant myxoid stroma of UES accounts for the attenuation lower than that of soft tissue on CT scans and the high signal intensity on T2 weighted MRI, as was in our case (7). Moon et al. (8) have also described the discrepancy of internal architecture on US and CT, as an important characteristic of UES. Therefore, a diagnosis of UES is strongly suggested by the presence of a large hepatic lesion that has an apparent cystic appearance on CT and MRI but a largely solid appearance on US images. The use of delayed -phase contrast-enhanced CT and MRI is also demonstrated as useful because these techniques reveal tumoral enhancement and help to exclude

halefoglu-.indd 250

JBR–BTR, 2014, 97 (4)

purely cystic lesions in differential diagnosis (12). It has been known that the clinical outcome of UES is poor (4). However, many authors suggest that multiagent, adjuvant or neo-adjuvant chemotherapy followed by resection offers the best long-term results and possibly a cure (13). Patients who underwent complete resection followed by adjuvant chemotherapy had significantly better survival rates compared with patients who underwent surgical resection alone. In addition, better survival was reported with intensive adjuvant therapy, even when there was residual tumor after radical resection (14). There have been three reports of liver transplantation for UES in children (15, 16, 17), however the use of liver transplantation for primary hepatic sarcoma in adults is controversial. In conclusion, we hereby presented a 7 year-old male child with UES of the liver which was initially misdiagnosed and treated as hydatid cyst. Although the differential diagnosis with imaging modalities seems to be challenging between these two entities, awareness about the paradoxical imaging findings (i. e. solid appearance on US vs. cystic appearance on CT and MRI) is crucial, because early correct diagnosis and curative surgery are mandatory for the favorable outcome of these tumors. References 1. Sodhi K.S., Bekhitt E., Rickert C.: Paradoxical hepatic tumor: Undifferentiated embryonal sarcoma of the liver. Indian J Radiol Imaging, 2010, 20: 6971. 2. Joshi S.W., Merchant N.H., Jambhekar N.A.: Primary multilocular cystic undifferentiated (embryonal) sarcoma of the liver in childhood resembling hydatid cyst of the liver. Br J Radiol, 1997, 70: 314-316. 3. Wei Z.G., Tang L.F., Chen Z.M., Tang H.F., Li M.J.: Childhood undifferentiated embryonal liver sarcoma: Clinical features and immunohistochemistry analysis. J Pediatr Surg, 2008, 43: 1912-1919. 4. Stocker J.T., Ishak K.G.: Undifferen tiated (embryonal) sarcoma of the liver: report of 31 cases. Cancer, 1978, 42: 336-348.

5. Pachera S., Nishio H., Takahashi Y., et al.: Undifferentiated embryonal sarcoma of the liver: case report and literature survey. J Hepatobiliary Pancreat Surg, 2008, 15: 536-544. 6. Sakellaridis T., Panagiotou I., Georgantas T., Micros G., R ontogianni D., Antiochos C.: Undifferentiated embryonal sarcoma of the liver mimicking acute appendicitis. World J Surg Oncol, 2006, 4: 9. 7. Buetow P.C., Buck J.L., PantongragBrown L., et al.: Undifferentiated embryonal sarcoma of the liver: pathological bases of imaging findings in 28 cases. Radiology, 1997, 203: 779783. 8. Moon W.K., Kim W.S., Kim I.O., et al.: Undifferentiated embryonal sarcoma of the liver: US and CT findings. Pediatr Radiol, 1994, 24: 500-503. 9. Charfi S., Ayadi L., Toumi N., et al.: Cystic undifferentiated sarcoma of liver in children: a pitfall diagnosis endemic hydatidosis areas. J Pediatr Surg, 2008, 43: E1-4. 10. Faraj W., Mukherji D., Majzoub N.E., Shamseddine A., Shamseddine A., Khalife M.: Primary undifferentiated embryonal sarcoma of the liver mistaken for hydatid disease. World J of Surg Oncol, 2010, 8: 58. 11. Yoon J.Y., Lee J.M., Kim do Y., et al.: A case of embryonal sarcoma of the liver mimicking a hydatid cyst in an adult. Gut and Liver, 2010, 4: 245-259. 12. Psatha E.A., Semelka R.C., Fordham L., Firat Z., Woosley J.T.: Undifferentiated (embryonal) sarcoma of the liver (USL): MRI findings including dynamic gadolinium enhancement. Magn Reson Imaging, 2004, 22: 897-900. 13. Baron P.W., Majlessipour F., Bedros A.A., et al.: Undifferentiated embryonal sarcoma of the liver successfully treated with chemotherapy and liver resection. J Gastrointest Surg, 2007, 11: 73-75. 14. Newmann K.D., Schisgall R., Reaman G., Guzzetta P.C.: Malignant mesenchymoma of the liver in children. J Pediatr Surg, 1989, 24: 781-783. 15. Kelly M.J., Martin L., Alonso M., Altura R.A.: Liver transplantation for relapsed undifferentiated embryonal sarcoma in a young child. J Pediatr Surg, 2009, 44: E1-3. 16. Dower N.A., Smith L.J.: Liver transplantation for malignant liver tumors in children. Med Pediatr Oncol, 2000, 34: 132-135. 17. Okajima H., Ohya Y., Lee K.J., et al.: Management of undifferentiated sarcoma of the liver including living donor liver transplantation as a back up procedure. J Pediatr Surg, 2009, 44: E33-38.

5/08/14 14:05

JBR–BTR, 2014, 97: 251-253.

Appearance and vanishing of a spinal intradural arachnoid cyst after multiple epidural corticosteroid injections. A spontaneously resolving cause of symptomatic lumbar stenosis P. Mailleux1, G. Milbouw², G. Koerts², P. Verbeek³, X. Willems³ Background: Spinal intradural arachnoid cyst is a rare lesion, sometimes acquired. Purpose: To describe the appearance and later spontaneous disappearence of a lumbar intradural arachnoid cyst, following perineural corticoid injections. Method: Review of the clinical data and imaging, with final spontaneous return to initial state. Discussion: Atypical intradural arachnoid cysts can be related to perineural injections and can cause symptoms of spinal stenosis. Its spontaneous vanishing is a very rare event, up to now unreported. Key-word: Arachnoid, cysts.

Intradural spinal arachnoid cysts are rare, sometimes symptomatic with radiculopathy, cord compression or symptoms of canal stenosis (1-4). They can be incidental findings on MRI. The majority lay dorsally (80%), only 20% are ventrally located. Only 5% occur in the lumbar region, most being thoracic (80%) with 15% in the cervical spine. Their etiology remains uncertain. Most are thought to be congenital, some being related with the development of arachnoiditis, or following surgical or percutaneous dural approach. Case report The patient is a 80 years old female patient with chronic cervical and lumbar pain, and non specific irradiation in both lower legs, more painful on the left side. She consulted multiple physicians and multiple L4-L5 and L5-S1 intra-articular corticosteroid injections were performed during the last 10 years in various hospitals, as well as epidural corticosteroid injections, while the diagnosis of “restless legs syndrome” was formerly proposed by neurologist. MRI performed in November 2011 (Fig. 1A) showed chronic lumbar discopathies, and normal distribution of the roots in a large lumbar thecal sac. Two more periradicular epidural corticoid injections close to the intraforaminal left L5 root were performed in January and February 2012 (Fig. 2). In March 2012, the patient was not able to walk more than one hundred meters, which was diagnosed as a possible “neurological claudication”.

Fig. 1. — A. Sagittal T2 MRI of the lumbar spine (November 2011): multiple discopathies, no stenosis. B. Sagittal T2 MRI of the lumbar spine (April 2012): lumbar nerve roots pushed anteriorly by a cystic lesion.

From: 1. Dept of Medical Imaging, Clinique St Luc, Bouge, 2. Dept of Neurosurgery, Regional Hospital Center, Namur, 3. Dept of Medical Imaging, Clinique et Maternité Sainte Elisabeth, Namur, Belgium Address for correspondence: Dr P. Mailleux, M.D., Dpt Of Medical Imaging, Clinique St Luc, rue St Luc 8, 5004 Bouge, Belgium. E-mail: p.mailleux@skynet.be

Mailleux et al.indd 251

Fig. 2. — February 2012: perineural infiltration close to left L5 nerve root (arrows pointing to the needle).

5/08/14 14:13

252

JBR–BTR, 2014, 97 (4)

Fig. 3. — A to F: axial T2 slice of the lumbar spine. Posterior cystic lesion pushes the nerves anteriorly and causes severe spinal stenosis. Loculations are well seen at the lower level behind disc L5-S1.

Electromyogram was unchanged, showing signs of chronic light neurogenic involvement in the left L5 root territory. Another lumbar MRI in April 2012 showed unchanged chronic discal alterations and all the roots pushed anteriorly at the L3-L4 and L4-L5 levels by a large intradural multi loculated cyst (Fig. 1B, Fig. 3). In con sideration to the patient age, no surgery was proposed and absten tion of anymore epidural injection was advocated. Patient inability to walk more than a few meters subsided and a control MRI three months later (Fig. 4) showed complete disappearance of the cystic lesions in the lumbar dural space and return to normal posterior location of the roots in the thecal sac. Discussion

Fig. 4. — A (July 2012): Sagittal and axial T2 images showing normal dorsal position of the roots in the thecal sac, with regular distribution of the roots.

Mailleux et al.indd 252

The first lumbar MRI of the patient, at the age of 79 years shows only degenerative disc disease, without stenosis or signs of arachno iditis. She received two epidural corticoidinjections afterwards, close to the left L5 root, in the foramen

5/08/14 14:13

ARACHNOID CYST AFTER MULTIPLE EPIDURAL CORTICOSTEROID INJECTIONS — MAILLEUX et al

(Fig. 2). On the second lumbar MRI (Fig. 1B, 3B), the roots at the L3-L4 and L4-L5 levels are compressed anteriorlyby a large intradural arachnoid cyst which extends on 10 cms from the L1-L2 level superiorly to lower than the L5-S1 level and appears multiloculated. Inferiorly located images (Fig. 3E and 3F) show the caudal extension of the cyst, displacing the S1 and S2 roots. The T2 signal intensity in the cyst is higher than in the CSF around the roots (Fig. 3B), which can probably be explained by a lesser transmission of the arterial pulsation of the arteries in the cyst compared to the “normal” thecal sac (5), a kind of “reversed flow void phenomenon”. Due to the old age of the patient, a surgical treatment was not proposed and the patient symptoms improved with the ability to perform longer walk, but with persistent lumbar pain. And no cyst could be seen the July 2012 lumbar MRI : the lumbar roots lay dorsally, without clumping or real sign of arachnoiditis (Fig. 4). Most spinal intradural arachnoid cysts are thought to be congenital, some being related to arachnoid adhesionsdeveloping secondary to inflammation, which may arise from infection, hemorrhage, or an iatrogenic cause such as injected contrast

Mailleux et al.indd 253

media or anesthetics (6). A symptomatic lumbar arachnoid cyst appearing a few months after a normal myelography was reported, supporting evidence for the traumatic etiology of some spinal intradural arachnoid cyst (7). Our case seems to comfort the hypothesis, with the cyst appearing just a few weeks after two periradicular injections. Its appearance is different from the classic globular form, with some septations well seen on Fig. 3D. On the other hand, spontaneous vanishing of an arachnoid cyst seems to be very rare: just a few cases have been reported in the head (8-9), but not in the spine. One possible hypo thesisis that the needle (or the corticosteroid injected) during the perineural injection could have caused a dural lesion with a “valve” effect, dissecting the dura and leading to the multiloculated appearance of the cyst, but that healed after a few months, with regression of the cyst. It is probably the first case of spontaneously disappearance of an spinal intradural arachnoid cyst. References 1. Robinson Y., Reinke M., Hascht-

mann D., Ertel W., Heyde C.E.: Spinal extradural meningeal cyst with spinal

3. 4.

253

stenosis. Spinal Cord, 2006, 44: 457460. Nabors M.W., Pait T.G., Byrd E.B., et al.: Updated assessment and current classification of spinal meningeal cysts. J Neurosurg, 1988, 68: 366-377. Wang M.Y., Levi A.D., Green B.A.: Intradural spinal arachnoid cysts in adults. Surg Neurol, 2003, 60: 49-55. Osenbach R.K., Godersky J.C., Traynelis V.C., Schelper R.D.: Intra dural extramedullary cysts of the spinal canal: clinical presentation, radiographic diagnosis, and surgical management. Neurosurgery, 1992, 30: 3542. Brooks M.L., Jolsez F.A., Patz S.: MRI of pulsatile CSF motion within arachnoid cysts. Magnetic resonance imaging, 1988, 6: 575-584. Hughes G., Ugokwe K., Benzel E.C.: A review of spinal arachnoid cysts. Cleveland Clinic J Med, 2008, 75: 311315. Kriss T.C., Kriss V.M.: Symptomatic spinal intradural arachnoid cyst development after lumbar myelography. Case report and review of the literature. Spine, 1997, 122: 568-572. Weber R., Lumenta C., Lenard H.G.: Spontaneous regression of a temporal arachnoid cyst. Childs Nerv Syst, 1991, 7: 414-415. Cokluk C., Senel A., Celik F., Ergür H.: Spontaneous disappearance of two asymptomatic arachnoid cysts in two different locations. Minim Invasive Neurosurg, 2003, 46: 110-112.

5/08/14 14:13

JBR–BTR, 2014, 97: 254-258.

HEPATIC ANGIOSARCOMA OCCURRING 65 YEARS AFTER THORIUM DIOXIDE (THOROTRAST) EXPOSURE: IMAGING, SURGICAL AND HISTO PATHOLOGIC FINDINGS OF A HISTORICAL CASE B. Coulier1, F. Pierard2, I. Gielen3, Ph. Maldague4 We report the CT, surgical and histopathologic findings of a rare case of Hepatic Angiosarcoma (HAS) diagnosed in a 85-year old women 65 years after Thorotrast (Th²³²) exposure for angiography. At the early arterial phase of dynamic MDCT, peripheral curvilinear and central nodular puddling of contrast produced in the 8 cm tumor. Then progressive contrast filling of the tumor was observed on the delayed scans. Associated pathognomonic signs related to previous Th²³² exposure were also found comprising diffuse intrahepatic reticular bands of calcifications, numerous calcified epigastric lymph nodes and a calcified shrunken spleen. Emergency laparotomy was performed because of associated hemoperitoneum. With a delay of 65 years after Thorotrast exposure, this historical case probably represents, to our knowledge, the most delayed presentation of Th²³² related HAS ever published. Key-words: Liver neoplasms – Thorium oxide.

Hepatic Angiosarcoma (HAS) is the more common mesenchymatous liver tumor. Nevertheless it remains rather rare and accounts for less than 2% of primary hepatic tumors (1, 2). Clinical diagnosis is usually difficult because symptoms and signs are non specific (2, 3). Moreover HAS is also difficult to differentiate from other hepatic tumors even with modern imaging techniques. As a rule the tumor progresses rapidly and has a poor prognosis (4). We present a rare case of HAS fortuitously diagnosed in an 85-year-old woman. The diagnosis was prompt for several reasons: the patient presented with spontaneous hemoperitoneum – a classical complication of HAS precipitating emergency imaging and surgery –, the MDCT findings were rather typical of a vascular tumor and finally pathognomonic CT signs related to previous Thorotrast (Th²³²) exposure – known as the most known iatrogenic cause of HAS – were associated. The history of the problems associated to the use of Thorotrast (TH²³²) and the imaging features of HAS are briefly remembered (4-7). With a delay of 65 years after Thorotrast exposure, this “historical” case probably represents, to our knowledge, the most delayed presentation of Th²³² related HAS ever published.

state, macrocytic anemia and left subcostal and epigastric pain exacerbated by meals. Laboratory tests at admission confirmed the anemia with hemoglobin level at 81 g/L (normal range 120160 g/L), a mild inflammatory syn-

drome with C-reactive protein level at 45 mg/L (normal value < 5 mg/L) and alteration of the hepatic function with SGOT at 36 U/L (normal value < 35 U/L), SGPT at 17 U/L (normal value < 31 U/L), GGT at 121 U/L (normal value < 36 U/L), LDH at 368 U/L

Fig. 1. — Hepatic MDCT axial views obtained during arterial phase (A) (= 30 sec after contrast injection), mixed arterio-portal semi-delayed phase (B) (= 120 sec after contrast injection) and delayed phase (C) (= 180 sec after injection). The liver appears diffusely heterogeneous with diffuse fine lace-like reticular bands Case report of calcifications (small black arrows). An 8 cm hypodense heterogeneous mass if found in the S2 & S3 segments (white arrows) A 85-year-old woman was admitas clearly confirmed on the arterial coronal oblique view (D) ted in the department of gastroenwhere the umbilical fissure is clearly delineated (grey arrow). terology for alteration of the general During the arterial phase (A, D) atypical and unusual diffuse irregular curvilinear peripheral and nodular central puddling of contrast enlightens the mass (small black arrows). Progressive filling produces durFrom: Department of 1. Diagnostic Radiology, 2. Visceral Surgery, 4. Gastroenterology, ing the intermediate time (B) and appears Clinique St Luc, Bouge (Namur), 3. Institute of Pathology and Genetics, Loverval, maximal on the delayed phase (C). Only Belgium. two ovoid areas remain hypodense probAddress for correspondence: Dr B. Coulier, M.D., Department of Diagnostic Radiology, ably corresponding to cystic and/or neClinique St Luc, Rue St Luc 8, 5004 Bouge (Namur) Belgium. crotic foci (black stars). E-mail: bcoulier@skynet.be

Coulier et al.indd 254

7/08/14 08:21

HEPATIC ANGIOSARCOMA OCCURRING 65 YEARS AFTER THORIUM DIOXIDE EXPOSURE — COULIER et al

255

A C

Fig. 2. — Thick coronal oblique maximal intensity projection (MIP) view (A), Volume Rendering (VR) view (B) and very thick average MPR view (C) illustrate very pathognomonic findings consistent with prior Thorotrast (Thorium dioxide or Th²³²) exposure: lace-like reticular calcifications within the hepatic parenchyma (black stars), multiple extremely dense calcified epigastric lymph nodes (black arrow) and a calcified shrunken spleen (white arrow). Thorotrast is displaced circumferentially by the tumor (small white stars) and is concentrated in its periphery in a capsule like fashion (little white stars). Axial view of the pelvic area (D) shows hyperdense hemorrhagic ascitic fluid (white star).

(normal value < 250 U/L), and ALP at 246 U/L (normal range 35-105 U/L). Gastroscopy was normal but abdominal ultrasound (not illustrated) revealed an 8 cm solid mass in the left hepatic lobe. Multiphasic contrast enhanced abdominal MDCT was performed (Fig. 1). An 8 cm hypodense tumoral mass was confirmed in the S2 & S3

B Fig. 3. — Intra-operative view (A): diffuse intraperitoneal free hemorrhage is visible and more than one liter of fresh blood was aspired. The entire surface of the liver has a pale tan reticulated pattern where the Thorotrast accumulated and caused fibrosis (white arrows). The S2 and S3 tumorous segments appear extremely nodular, congestive (black arrow) and numerous superficial erosions are spontaneously bleeding. Gross anatomy (B): at opening the degenerated left lobe has an extremely irregular spongious appearance constituted by an anarchic network of numerous irregular necrotic and hemorrhagic cavities.

A B Fig. 4. — Histopathology (A): accumulation of a birefrengent Thorotrastic granular material (white arrows) is seen within endothelial neoplastic cells (hematoxylin and eosin staining; magnification of 100x). B. The typical angiosarcomatous aspect (black star) is constituted by a network of immature anastomotic vascular spaces boarded by very atypical hyperchromatic endothelial cells. The spaces contain red and hematopoietic cells. White star = normal liver.

Coulier et al.indd 255

7/08/14 08:21

256

segments. Diffuse irregular curvilinear peripheral and nodular central puddling of contrast enlightened the mass during the arterial phase. Then progressive filling produced during the portal phase and appeared maximal on a delayed phase. Only two ovoid areas remained hypodense probably corresponding to cystic and/or necrotic foci. These characteristics were considered compatible with a vascular tumor. Associated but fundamental findings in terms of specific diagnosis were simultaneously found in the epigastric area comprising diffuse intrahepatic fine lace-like reticular bands of calcifications, the presence of multiple extremely dense calcified epigastric lymph nodes and finally a calcified shrunken spleen. These pathognomonic findings were extremely similar to those reported in the literature in patients having previously being exposed to Thorotrast (Th²³²). Further interrogation of the old woman revealed a previous history of cerebral arteriography performed in 1948 at the age of 20 years. This arteriography had been performed with Thorotrast to investigate tinnitus. The decisive CT finding justifying an emergency laparotomy was the presence fresh hemorrhagic ascites in the pelvic cavity (Fig. 2). The final preoperative radiologic diagnosis was that of a bleeding hepatic angiosarcoma (HAS) that had developed 65 years after Thorotrast exposure. The clinical state of the old woman suddenly deteriorated with discomfort, hypotension and an acute recrudescence of epigastric pain. Emergency laparotomy found more than one liter of fresh blood in the peritoneum. The entire surface of the liver – where Thorotrast had accumulated and had induced fibrosis – had a pale tan reticulated pattern (Fig. 3). The S2 and S3 tumorous segments were diffusely nodular and congestive with numerous spontaneously bleeding superficial erosions. At opening the tumor had an extremely irregular spongious appearance constituted by an anarchic network of numerous irregular necrotic and hemorrhagic cavities. Histopathology (Fig. 4) revealed accumulation of birefrengent thoro trastic granular material within neoplastic endothelial cells. The typical angiosarcomatous aspect was confirmed and constituted by a network of immature anastomotic vascular spaces boarded by very atypical hyper chromatic endothelial cells. The immediate postoperative period was uneventful but unfortunately

Coulier et al.indd 256

JBR–BTR, 2014, 97 (4)

the general state of the old women altered rapidly. She experienced recurrent episodes of ischemic strokes and died one month later. Discussion Thorotrast was the trade name of an X-ray contrast medium used worldwide from about 1928 to 1955 (4-7). The preponderant form of administration was intra-arterial injection mostly for cerebral angiography, but this contrast medium was also used for ventriculography, hepatospleno-portography, pyelography, urethrography, hysterosalpingography and instillation of anatomic cavities comprising paranasal sinuses. It consisted of a 25% colloidal solution of thorium dioxide (Th²³²). After intravascular injection Th²³² aggregates were stored lifelong in the reticulo endothelial system (4-7). Approximately 70% of the medium was taken by the liver, 20% by the spleen and the remainder by the bone marrow and lymph nodes causing a chronic exposure to alpha-(90%), beta- (9%) and gamma- (1%) rays especially in these organs. The biological half-time was of 400 years (4-7). Approximately ten years after its introduction, first reports of possible carcinogenic effects, especially tumors formation in the liver, were published in the international literature. Despite these publications, the use of Th²³² increased, because of the lack of acute toxicity and excellent radiological results compared with other contrast media. With time, the carcinogenic effects of Th²³² became increasingly clear and numerous cases of Th²³²-related malignancies were reported, especially malignant hepatic tumours, such as hepatocellular carcinoma, cholangiocarcinoma and hemangiosarcoma (HAS) (2-7). Th²³² was abandoned following a reportby MacMahon et al (8) of HAS attributed to Thorotrast exposure in 1947 (6-8). Since about 1950, various national epidemiologic studies, sometimes extending over very long periods, undertook observation of large series of exposed patients and compared them with cohorts of control patients. The purpose was the study of the long term effect of Th²³² in the depository organs (5, 9). The main related reported diseases and causes of death were malignant primary liver tumors (HAS, hepatoma, cholangiocarcinoma), cirrhosis of the liver, blood diseases comprising anaplastic anemia, thrombocy-

topenic anemia, hemolytic anemia, myelofibrosis and other neoplastic diseases comprising cancers of the extrahepatic bile duct, pancreas, oesophagus, larynx, non-hodgkin’s lymphoma, bone sarcomas, plasmocytomas and mesothelimas (5-7). Various rare cases of transitional cell carcinoma or squamous cell carcinoma due to suburothelial thorium deposition – thorotrast kidney – have been described in patient who had undergone retrograde pyelography with Thorotrast (10). Statistical analysis showed that the incidences of these disorders were significantly higher in the exposed patients than in the controls. The lifespan of Thorotrast administered persons decreased with the amount of Th²³² injected. A clear mean dose rate effect relationship exists: the tumor frequency depends on the time of exposure or the cumulative dose to the liver respectively and not on the age at injection (4-5). Three criteria must be met before implicating Th²³² as a cause of neoplasia: the Th²³² must be present in vicinity of the tumor, the latent period must be sufficiently long (average 20 years) and the dose must have been sufficiently high (6). CT findings of Th²³² deposition are extremely pathognomonic. Typically high-density deposits are seen in the liver, spleen and lymph nodes and the atrophy of the spleen due to fibrosis is also a typical sign (2-4). Deposition in bone marrow is also described (4). Beside the common hepatocellular carcinoma, more rare malignant tumours of the liver are occasionally seen in daily clinical practice. Although benign vascular tumors of the liver are extremely common (hemangioma being the most familiar) malignant vascular tumors (HAS, epithelioid hemangioendothelioma and Kaposi sarcoma) are very rare (2). Although it is the more common mesenchymatous liver tumor occuring more frequently than fibrosarcoma, malignant fibrohistyocytoma and leiomyosarcoma, HAS accounts for less than 2% of primary hepatic tumors, with an estimated frequency of 0.14 to 0.25 per million (1-4). It is more common in late adulthood (6th-7th decade) and in males (ratio 4:1). Etiological factors in HAS may be environmental or occupational exposure to carcinogens comprising thorium dioxide (Th²³²), polyvinyl chloride, arsenic, inorganic copper and anabolic steroids (1). In all cases of environmental exposure, a prolonged latency period has been

7/08/14 08:21

HEPATIC ANGIOSARCOMA OCCURRING 65 YEARS AFTER THORIUM DIOXIDE EXPOSURE — COULIER et al

established of 20-30 years on average (6). Th²³² is the most known iatrogenic cause of HAS the liver, 7-10% of HAS being Th²³² related (2, 4). HAS is also associated with hemochromatosis and von Recklinghausen disease (1, 3). However, most of HAS (75%) occur either in the absence of know risk factors or with cirrhosis of the liver (1, 2). The diagnosis of HAS is often performed too late due to nonspecific symptoms comprising abdominal pain, weakness, alteration of the general state (2, 3). Ascites and jaundice may be seen. Hemorrhagic ascitis is common and spontaneous hemoperitoneum and splenic metastases also occur (2, 3). In HAS vessels are lined with malignant endothelial cells (2). The tumor cells also grow along preformed vascular channels, particularly the sinusoids, and may form solid nodules consisting of malignant spindle cells or cavitary spaces lined with tumor cells. In cases related with Th²³² the tumor grows in an infiltrating pattern and contains some Th²³² but the majority of the product however is displaced circumferentially by the tumor and is concentrated in the periphery of the HAS in a capsule like fashion (2, 3). This situation was found in the reported case (Fig. 1). Additionnally in cases exposed to Th²³² the entire surface of the liver – as also typically observed in our case (figure 3) – has a pale tan reticulated pattern where the Th²³² accumulated and produced secondary fibrosis. In the absence of Th²³² exposure, The CT appearance of HAS is consis tent with a vascular tumour, in which two predominant growth patterns can be seen: multinodular lesions or a predominantly large solitary mass (1, 2, 4). These masses generally appear spontaneously hypo attenuating on native CT (2, 11). Hyperattenuating areas or areas of very low attenuation near that of fluid may also be found respectively corresponding to fresh hemorrhage or old hemorrhage or necrosis (2). On the dynamic contrast-enhanced CT images and when it appears as a large mass, HAS may show heterogeneous enhancement during the arterial phase. At delayed imaging, there is progressive diffuse enhancement of the lesion compared with that of the early phase images. This enhancement pattern on dynamic contrast enhanced images merely mimics that of a cavernous hemangioma, a benign tumor which is

Coulier et al.indd 257

finally 10.000 times more common than HAS ! Histopathologically multiple vascular channels that are separated by fibrous septa are similar in both tumors (1, 2, 11, 12). During the hepatic parenchymal phase or delayed post-contrast scans, the entire mass or at least some of its areas become isodense with normal liver (11). When typical central necrosis or hemorrhage is present substantial and prolongated peripheral enhancement may appears and may mimic a very large benign angioma in which incomplete contrast filling is a rule (2, 7). Recent reports have demonstrated some distinctive aspects of HAS at biphasic imaging when compared with classical hemangioma (3, 11): HAS shows more heterogeneous and persistent enhancement that may be less than that of the aorta or hepatic artery, while typical hemangioma show progressive centripetal nodular enhancement that is of similar density of the contrast opacified blood in the aorta or the hepatic artery during all phases of imaging (3). Moreover some cases show not only peripheral curvilinear puddles of contrast but also multiple irregular central flecked enhancements – as present in the reported case (Fig. 1) – on early contrast-enhanced CT (11). When not related to Th²³² exposure, HAS, whatever presenting as multiple nodules or as a single mass, often cannot be readily distinguished from hypervascular metastases (such as from neuroendocrine tumors) and hepatocellular carcinoma (3). All of these tumors may demonstrate internal hemorrhage and heterogeneity, in addition to early and heterogeneous enhancement. In contrast to hepatocellular carcinoma, however, HAS demonstrates continuing, progressive enhancement on delayed-phase images. Splenic metastases, associated hematologic abnormalities and the lack of cirrhosis or elevated alpha-fetoprotein may also suggest HAS rather than of hepatocellular carcinoma. The differential diagnosis also concerns other vascular tumors comprising hemangioma, epithelioid hemangioendothelima, cholangiocarcinoma and hepatoblastoma (13). When Th²²² exposure is recognized other types of Th²³² related neoplasms have to be considered comprising hepatocarcinoma (HCC) but also intrahepatic-cholangiocarcinoma (2). The presence of splenic metastases and/or hemoperitoneum are suggestive of HAS. On the con-

257

trary an elevated alpha fetoprotein level is rather suggestive of HCC. In general HCC show a rapid global arterial phase enhancement depending on its vascularity, which rapidly decreases to the attenuation value of normal liver (8). In contrast to nonTh²³² related cases which are preponderantly located in the hepatic hilum, Th²³² related cholangiocarcinoma are preponderantly of the peripheralmiddle type, in which the tumor is located in the periphery to middle portion of the liver (14). Peripheral cholangiocarcinoma classically manifests as a large, well-defined hepatic mass with lobulated margins and peripheral rim enhancement (15). Moreover cholangiocarcinoma frequently demonstrates intrahepatic ductal dilatation (2, 7). The prognosis of HAS is very poor, almost every patient dying within one year of diagnosis whatever the protocol of chemotherapy used. Attempted radical resections in case of localized disease are also very disappointing with fast relapses of the tumor. Even liver transplantation does not seem feasible in this disease (4). Conclusion Rare cases of Thorotrast-related HAS will continue to occur more and more sporadically and the disease will disappear in a few years. The reason is the convergence between a sometimes very long latency after exposure and the fact that the patients are today extremely old – if not already died from various diseases or simply from old age. The use of Thorotrast having been prohibited since 1950, even patients aged 20 to 30 years old at that time are in fact now aged of 83 to 93 years. Nevertheless the reported case remains didactic for young radiologists to be aware of the history of Thorotrast-related hepatic neoplasms. Moreover the MDCT findings of HAS reported in this report are also typical and are independent of a Thorotrast exposure. References 1. Heo S.H., Jeong Y.Y., Shin S.S.,

Chung T.W., Kang H.K.: Solitary small hepatic angiosarcoma: initial and follow-up imaging findings. Korean J Radiol, 2007, 8: 180-183. 2. Buetow P.C., Buck J.L., Ros P.R., Goodman Z.D.: Malignant vascular tumors of the liver: radiologic-pathologic correlation. Radiographics, 1994, 14: 153-166. 3. Koyama T., Fletcher J.G., Johnson C.D., Kuo M.S., Notohara K., Burgart L.J.:

7/08/14 08:21

258

Primary hepatic angiosarcoma: findings at CT and MR imaging. Radiology, 2002, 222: 667-673. 4. van Kampen R.J., Erdkamp F.L., Peters F.P.: Thorium dioxide-related haemangiosarcoma of the liver. Neth J Med, 2007, 65: 279-282. 5. van Kaick G., Wesch H., Lührs H., LiebermannD., Kaul A.: Neoplastic diseases induced by chronic alpha-irradiation -epidemiological, biophysical and clinical results of the German Thorotrast Study. J Radiat Res, 1991, 32: 20-33. 6. Weber E., Laarbaui F,. Michel L., Donckier J.: Abdominal pain: do not forget Thorotrast! Postgrad Med J, 1995, 71: 367-368. 7. Azodo M.V., Gutierrez O.H., Greer T.: Thorotrast-induced ruptured hepatic

Coulier et al.indd 258

JBR–BTR, 2014, 97 (4) angiosarcoma. Abdom Imaging, 1993, 18: 78-81. 8. Macmahon H.E., Murphy A.S., Bates M.I.: Endothelial-Cell Sarcoma of Liver Following Thorotrast Injections. Am J Pathol, 1947, 23: 585611. 9. Mori T., Kato Y.: Epidemiological, pathological and dosimetric status of Japanese thorotrast patients. J Radiat Res, 1991, 32: 34-45. 10. Oyen R.H., Gielen J.L., Van Poppel H.P. et al.: Renal thorium deposition associated with transitional cell carcinoma: radiologic demonstration in two patients. Radiology, 1988, 169: 705-707. 11. Yu R., Zhang S., Hua J.: Hepatic angiosarcoma: CT findings. Chin Med J (Engl), 2003, 116: 318-320.

12. Peterson M.S., Baron R.L., Rankin S.C.: Hepatic angiosarcoma: findings on multiphasic contrast-enhanced helical CT do not mimic hepatic hemangioma. AJR, 2000, 175: 165-170. 13. Rademaker J., Widjaja A., Galanski M.: Hepatic hemangiosarcoma: imaging findings and differential diagnosis. Eur Radiol, 2000, 10(1): 129-133. 14. Kojiro M., Sugihara S., Ito Y. et al.: Pathomorphological study of thoro trast-related intrahepatic cholangiocarcinoma – a comparison with nonthorotrast cases. Gan No Rinsho. Japan Journal of Cancer Clinics, 1986, 32: 349-355. 15. Han J.K., Choi B.I., Kim A.Y. et al.: Cholangiocarcinoma: pictorial essay of CT and cholangiographic findings. Radiographics, 2002, 22: 173-187.

7/08/14 08:21

JBR–BTR, 2014, 97: 259-261.

Fibrous pseudotumor of the tunica vaginalis: contrast enhanced sonography with pathologic correlation T. Puttemans1, M.I. Ingabire1, J.L. Jorion2, A.P. Draguet3 We report the case of a 28-year-old man who presented at the emergency department with recent left painful scrotal swelling, without history of genitourinary infection or trauma. On physical examination, left scrotal swelling with nodular palpation was noted. Contrast enhanced sonography demonstrated nodular vascularized thickening of the tunica vaginalis. Surgical exploration revealed multiples solid nodules of the vaginal wall, with, at frozen section analysis, fibroblastic tissue, vessels and chronic inflammation without malignity, suggestive of fibrous pseudotumor of the tunica vaginalis. We discuss the sonographic aspect of this rare entity and the difficulty to establish a diagnosis of benignity without surgical exploration. Key-word: Scrotum, US.

Scrotal fibrous pseudotumor is a rare tumor characterized by benign reactive fibroinflammatory tissue involving the paratesticular space, most frequently the tunica vaginalis (76%), less often the epididymis (10%), the tunica albuginea or the spermatic cord (1). Different sonographic aspects have been described, generally nonspecific and insufficient to avoid surgical exploration, required to rule out malignant processes. Case report A 28-year-old man presented at the emergency department with recent left painful scrotal swelling. No history of previous scrotal infection or trauma was noted. General physical examination and blood tests were normal (no inflammatory syndrome, C-reactive protein = 0,7 mg/dl). Scrotal examination performed by a senior urologist revealed left scrotal painful swelling with nodular palpation. Scrotal ultrasound (US) performed with a 9 MHz linear probe (Acuson S2000TM system from Siemens Medical Solutions) showed a multinodular, mild echoic, thickening of the tunica vaginalis (1,6 mm) associated with moderate anechoic hydrocele and normal testicle (Fig. 1A). No color Doppler (CD) signal was observed within the nodules (Fig. 1B). Epididymis had a normal US aspect as the right scrotum. Contrast enhanced sonography (CEUS) of the left scrotal wall, obtained after bolus intravenous injection of 4 ml

B Fig. 1. — A. Bmode ultrasound of the left scrotum showing multinodular thickening of the tunica vaginalis, surrounded by anechoic hydrocèle and normal testicle. B. Color Doppler sono graphy of the nodules showing the absence of signal.

From: 1. Department of Radiology, Clinique Saint-Pierre, Ottignies, 2. Department of Surgery, Clinique Saint-Pierre, Ottignies, 3. Department of Pathology, IPG, Gosselies, Belgium. Address for correspondence: Dr T. Puttemans, M.D., Department of Radiology, Clinique St-Pierre Ottignies, 1340 Ottignies, Belgium. E-mail: puttemans@clinique-saint-pierre.be

puttemans-.indd 259

5/08/14 14:30

JBR–BTR, 2014, 97 (4)

KLEUR

260

Fig. 3. — Gross specimen before excision showing multiple solid nodules of the vaginal wall, arranged in bunch of grapes. Fig. 2. — Contrast enhanced sonography showing homogeneous enhancement of the nodules.

contrast media (SonovueR, Bracco), showed progressive, homogeneous, enhancement of the nodules, followed by a slow decrease of the signal intensity without washout aspect at the late time (Fig. 2). Surgical exploration revealed multiple solid nodules of the vaginal wall, arranged in bunch of grapes (Fig. 3). A fragment of one nodule was submitted to frozen section analysis which showed fibroblastic tissue, vessels and chronic inflammation without malignity. All the visible nodules were removed by the surgeon without touching the testicle. Diagnosis of fibrous pseudotumor was confirmed by histologic analysis showing normal mesothelial cells without atypia, proliferation of fibroblast cells mixed with collagen fibers surrounding vascular structures and focal inflammatory mononuclear cells (Fig. 4). Immunohistochemical staining on the spindle component was negative for calretinin, cytokeratin 5/6, cytokeratin AE1/AE3 and WT1. Discussion Reactive fibroinflammatory lesions of the paratesticular space, so called fibrous pseudotumor, is a rare entity, generally observed in the third decade (patients range from 16 to 75 years of age), very rarely in paediatric population (1, 2). Fibrous pseudotumor is a generic term proposed by Mostophy that includes a series of different name like inflammatory pseudotumor, nodular and diffuse fibrous proliferation, proliferative funiculitis, chronic prolifer-

puttemans-.indd 260

Fig. 4. — Microscopic section (hematoxylin and eosin x100) showing diffuse sclerous tissue with few fibroblast and cluster of inflammatory cells around a vessel.

ative periorchitis, reactive periorchitis, fibromatous periorchitis, fibrous mesothelioma, benign fibrous paratesticular tumor, nonspecific peritesticular fibrosis (3). The origin of this fibroinflammatory reaction is unknown and the pathogenesis remains obscure. It is frequently associated with history of prior infection or trauma but sometimes no apparent cause is found (3, 4). Schistosomia haematobium infection and HIV infection have also been associated with fibrous pseudotumor (5). Macroscopically, nodules have gray-white and fibrogelatinous cut appearance with a whorled pattern (2). Histologically, they contain hyalinized fibrous tissue with scattered collagen bundles and proliferative fibroblasts, some vascular channels and scattered inflammatory cells,

rare focal calcification and ossification. Myxoid change may be present (2, 6). Immunohistochemical staining of the fibrous pseudotumor is highly positive for vimentin, smooth muscle-specific actin, and common muscle actin and negative for calretinin, cytokeratin 5/6 and WT1, specific markers for mesothelioma. Necrosis and increased areas of mitosis and pleomorphism are absent (4). Clinical examination (palpation) usually reveals single or multiple, painless, firm nodule, frequently associated with hydrocele or hematocele. Sometimes, scrotal painful swelling is the only reason for consultation. Different US aspects of scrotal fibrous pseudotumor have been described in the literature, depending on the localization.

5/08/14 14:30

FIBROUS PSEUDOTUMOR OF THE TUNICA VAGINALIS — PUTTEMANS et al

The most frequent US aspect is, like in our case, multiple solid hyperechoic or hypoechoic small masses, attached to the inner part of the parietal layer of the tunica vaginalis, surrounded by hydrocele (7, 9, 10). The echogenicity of the nodules depends on the amount of collagen or fibroblast content. No spontaneous CD signal is visible in the nodules and testicle is normal. Sometimes, bigger solid heterogeneous masses unrelated to the epididymis are associated with shadowing and small amount of CD vascularity (8). In our case, no CD signal was spontaneously visible in the nodules. Nevertheless, CEUS demonstrated a progressive, homogeneous, enhancement of the nodules that is consistent with the presence of vessels, followed by a slow decrease of the signal intensity, without washout aspect. This type of enhancement is highly suggestive of a benign enhancement. To our knowledge, this is the first description of such an enhancement. Less frequently, fibrous pseudotumor of tunica vaginalis appears like a small solitary mass, containing eventual focal central or linear calcification, fixed or free within the vaginalis, also described then as scrotal pearl (2, 9, 10). When the albuginea is involved (differentiating between albuginea and visceral layer of tunica vaginalis is impossible), a round, hypoechoic, mass of variable size, is attached to the testicle, simulating a testicular neoplasm (11). The extratesticular development of the mass within the vaginal cavity is typical with usual hydrocele. One case of bilateral synchronous fibrous pseudotumor of the tunica albuginea has been described in a paediatric patient (12). Diffuse fibrous pseudotumor, previously named nodular fibrous periorchitis or fibromatous periorchitis, is a very rare form of diffuse peritesticular thickening of the tunica vaginalis with focal linear calcifications (13). Others very rare localization of fibrous pseudotumor have been described as hypoechoic mass in the epididymal tail or in paratesticular lymph nodes (14). Sonographic differential diagnostic for fibrous pseudotumor of the

puttemans-.indd 261

paratesticular space includes, in the most frequent multinodular presentation, malignant mesothelioma, fibrosarcoma, leiomyosarcoma or rhabdomyosarcoma, in the solitary nodule presentation, fibrous mesothelioma, fibroma of the tunics, leiomyoma, neurofibroma, idiopathic fibromatosis and in the epididymal presentation, adenomatoid tumor or adenocarcinoma (8, 10, 14). Few MRI descriptions of fibrous pseudotumor have been publishedshowing intermediate to low signal intensity on T1-weighted images, similar to the testis, low signal intensity on T2-weighted images and little or no gadolinium enhancement (15). Despite the relative typical CD signal and MRI appearance of multinodular fibrous pseudotumor, imaging is generally not sufficient to affirm benignity and surgical exploration is required to rule out malignant processes. Only histological analysis with immunohistochemical staining may affirm the benign lesions and exclude malignancy. However, because fibrous pseudotumor is a benign process, radical orchiectomy is not necessary and local excision of the mass is the treatment of choice for histologic study and correct diagnosis (7). CEUS is an interesting new imaging modality which seems to give supplementary argument for the benign nature of the lesions. Unfortunately, for instance, no publication exists in the literature concerning the enhancement of scrotal fibrous pseudotumor and future CEUS explorations are needed to confirm this hypothesis. In conclusion, we present CD signal and CEUS appearance of a rare paratesticular fibrous pseudotumor. The radiologist should know this rare entity and its benign nature. Establish a correct preoperative diagnostic of benignity remains difficult but is important for correct surgical management. References 1. Ulbright T.M., Amin M.B., Young R.H.: Miscellaneous primary tumors of the testis, adnexa, and spermatic cord. Hematopoietic tumors. Secondary tumors. In: Rosai J., ed. Atlas of tumor pathology. Tumors of the testis, adnexa, spermatic cord and scrotum.

261

Washington, DC: AFIP, 1999: 247253. 2. Seethala R.R., Tirkes A.T., Weinstein S., Tomaszewski J.E., Malkowicz S.B., Genega E.M.: Diffuse fibrous pseudotumor of the testicular tunics associated with an inflamed hydrocele. Arch Pathol Lab Med, 2003, 127: 742-744. 3. Mostophi F.K., Price E.B.: Tumors of the male gentital system. In: Atlas of tumor pathology, 2nd series, fascicle 8. Washington DC: Armed Forces Institute of Pathology, 1973: 151. 4. Parker P.M., Pugliese J.M., Allen R.C.: Benign fibrous pseudotumor of tunica vaginalis testis. Urology, 2006, 68: 427e17-e19. 5. NavaiN., Yap R.L., Gupta R., Fraser T.G., Gonzalez C.M.: Inflammatory pseudotumor of the testis: a novel presentation of acute retroviral syndrome. Int J Urol, 2005, 12: 424-426. 6. Park B.K., Kim S.H., Moon M.H.: Fibrous pseudotumor of the epi didymis: sonographic and pathologic correlation. Eur J Radiol, 2003, 46: 5355. 7. Grebenc M.L., Gorman J.D., Sumika F.K.: Fibrous pseudotumor of the tunica vaginalis testis: imaging appearance. Abdom Imaging, 1995, 20: 379-380. 8. Germaine P., Simerman L.P.: Fibrous pseudotumor of the scrotum. J Ultrasound Med, 2007, 26: 133-138 9. Garriga V., Serrano A., Marin A., Medrano S., Roson N., Pruna X.: US of the tunica vaginalis testis: anatomic relationships and pathologic conditions. Radiographics, 2009, 29: 20172032. 10. Yang D.M., Kim H.C., Lim S.J.: Sonographic findings of fibrous pseudotumor of the tunica vaginalis. JCU, 2012, 40: 252-254. 11. Woodward P.J., Schwab C.M., Sesterhenn I.A.: Extratesticular scrotal masses: radiologic-pathologic correlation. Radiographics, 2003, 23: 215240. 12. Kern S.Q., McMann L.P.: Bilateral fibrous pseudotumors of the tunica albuginea in a pediatric patient. J Pediatr Urol, 2012, 8: e1-e3. 13. White W.M., Hilsenbeck J., Waters W.B.: Fibromatous periorchitis of testis. Urology, 2006, 67: 623 e15-6. 14. Krainik A., Sarrazin J.L., Camparo P., A., Vincendeau S., Houlgatte Cordoliani Y.S.: Fibrous pseudotumor of the epididymis: imaging and pathologic correlation. Eur Radiol, 2000, 10: 1636-1638. 15. Kim W., Rosen M.A., Langer J.E., Banner M.P., Siegelman E.S., Ramchandani P.: US-MR imaging correlation in pathologic conditions of the scrotum. Radiographics, 2007, 27: 1239-1253.

5/08/14 14:30

JBR–BTR, 2014, 97: 262.

IMAGES IN CLINICAL RADIOLOGY An unusual presentation of a pelvic textiloma mimicking a tumor R. Kadi1, V. Massard², K. Vanden Houte³, C. Gerard², L. Divano1, J. Jani², M. Laureys1, E. Najar1, M. Cannie1

A 21-year-old female patient consulted because of increasing vague pelvic pain. In her past history, an appendectomy was performed 15 years earlier. Endovaginal ultrasound showed an enlarged uterus with a nodular contour confirming the multiple leiomyomas. In the right pelvis a heterogeneous hypo vascular soft tissue mass was noted suspected to be an ovarian mass or a sub serous leiomyoma (Fig. A). At MR imaging, a right para-uterine mass was shown, sticking to the normal right ovary, excluding its ovarian origin. The mass had a hypointens signal on T1WI, heterogeneous hypo intense on T2WI and was delineated by a thick capsule showing enhancement after contrast injection (Fig. B, C). It had an extentrisic mass effect on the cervical region and pushed the right ovary in an anteroposteriorily way. A pediculated fibroma was excluded because of its characteristics. At MRI we were unable to differentiate between an old postoperative hematoma and abscess, a leiomyoma with a cystic degeneration or a pseudo cystic mass. Therefore the pelvic mass was removed by laparoscopy. The surgery revealed a nodular mass with a thick fibro sclerotic capsule and some calcifications. After opening of the nodule it was found a retained surgical sponge revealing the diagnosis of a textiloma (Fig. D). Comment

A textiloma is a mass that is created by a retained surgical sponge or foreign body where due to reactive inflammatory changes with formation of a tick capsule. The reported prevalence varies between 1/100 to 1/5000 after a pelvic intervention and around 1/1000 to 1/1500 after an intervention on the digestive tract (1). Normally the diagnosis will be made in the postoperative period due to abdominal pain or septicemia with formation of an abscess and/or a fistula. Rarely, due to the highly variable clinical and imaging presentation of this type of mass, the diagnosis is made on biopsy only. Mainly in cases where there is an aseptic encapsulation of the retained surgical sponge, the patient will remain asymptomatic for many years and the diagnosis made be more difficult. In most cases a textiloma is easily diagnosed on a convenC tional plain X-ray or on CT scan, due to the pathognomonic presence of marking filaments within it and/or on CT the presence of calcifications in the capsule and/or air in the mass as typically described in the literature due to air trapping. Unfortunately, it is only since the last decade that these marking filaments are used in operative compresses. Moreover, it is less likely to be used in underdeveloped country. As a consequence, not all textilomas will appear with marking filaments and the diagnosis several years postoperative may be very difficult. Typically, on MRI a textiloma, in a chronic setting, shows a thick capsule that has sharp delineations and where the capsule has a heterogeneous hyperintens signal on T2WI while it is mostly hypointense on T1WI due to its granulomatous and fibrotic tissue. After intravenous injection of contrast agent only the periphery capsule will enhance. Obviously, D depending on the chemical content of the textiloma, it can have a varying intensity on mainly the T1WI (1). Differential diagnosis of textiloma can be made with a postoperative hematoma or abscess, a cystic degeneration of a leiomyoma showing a hyperintens signal on T2WI due to the cystisation of the leiomyoma, a pseudo cystic pelvic tumour or an atypical dermoid cyst. In our case, a dermoid was excluded because of its extra-adnexial localisation and the absence of fatty tissue inside the lesion, which can be seen on the T1WI sequence with fat suppression. The pitfall in our case was the absence of the marking filaments and the presence of multiple uterine leiomyomas. The diagnosis of a textiloma should be included in the differential diagnosis of any atypical mass in the pelvis when there is a previous history of surgery, even when the typical marking filaments are absent. Reference 1. O’Connor A.R., Coakley F.V., Meng M.V., Eberhardt S.C.: Imaging of retained surgical sponges in the abdomen and pelvis. AJR, 2003, 180: 481-489. Department of 1. Radiology, 2. Obstetrics and Gynecology, 3. Pathology of the University Hospital Brugmann, Brussels, Belgium.

image-kadi-.indd 262

5/08/14 14:32

JBR–BTR, 2014, 97: 263.

IMAGES IN CLINICAL RADIOLOGY Leiomyoma of the urinary bladder T. Dewaele1, L. D’Hooghe1, St. Weyers2, P. Devisschere1

A routine gynaecological examination in a 26-year-old woman revealed a palpable mass behind the pubic symphysis. Transvaginal ultrasound showed a solid mass of 5 cm between the vagina and the urinary bladder protruding into the bladder. A transvaginal biopsy was performed and the pathological report suggested a benign mesenchymal tumor, most likely a leiomyoma. MRI scan showed a well-circumscribed solid mass on the endovesical side of the right bladder wall. The lesion had a homogenous low signal intensity on T1WI (Fig. A) and a slightly heterogeneous medium signal intensity on T2WI (Fig. B). The lower portion of the lesion extended to the bladder neck (Fig. B, C). On cystoscopy the tumor was covered by intact mucosa and extended close to the right ureteral orifice. Since the patient had initially no complaints ultrasound monitoring was suggested. However, one year later, she developed increased urinary frequency, and it was decided to resect the leiomyoma. Laparotomic resection resulted in a well-circumscribed mass that could relatively easily be removed by enucleation. Laparotomy was preferred over laparascopic resection because of the proximity of the right ureteral orifice.The pathological examination confirmed the diagnosis of a leiomyoma. The postoperative period was uneventful and patient is doing well 15 months after surgery. Comment

Bladder leiomyomas are rare and account for less than 0.43% of all bladder tumors. Leiomyomas are classified in the category of benign mesenchymal tumors. They may arise throughout the genitourinary system, but the renal capsule is the most common location. Leiomyomas of the urethra are less common than leiomyomas of the bladder. Series of cases reported in literature show that up to half of the tumors are incidentally discovered during a routine pelvic examination or on imaging performed for other reasons. Recent reports have indicated a possible higher incidence in woman but the reason has not been elucidated yet. The association with female hormones has been suggested although the cases discovered incidentally may be higher in women due to the higher frequency of ultrasonographic and pelvic examinations in women. The clinical presentation of bladder leiomyomas is variable and ranges from asymptomatic to urinary infection, hematuria and obstructive or irritative symptoms. It is presumed that bladder leiomyomas produce symptoms primarily related to their location and secondly to their size. Bladder leiomyomas have been described to occur in endovesical, extravesical or intramural locations. Endovesical leiomyomas, the most common type, tend to cause symptoms in a greater degree than extravesical or intramural leiomyomas. Bladder leiomyoma occur as solitary tumors in most cases, nonetheless multicentric tumors have been described. Bladder leiomyomas are histologically identical to uterine leiomyomas and therefore they have the same characteristics on imaging. On ultrasonography a smooth, homogenous solid mass is usually demonstrated, although partially cystic appearing leiomyomas have been reported. On MRI leiomyomas usually show mediumsignal intensity on T1WI and homogenous low-signal intensity on T2WI. The optimal treatment for bladder leiomyoma has not been established yet. To date, surgical resection is the treatment of choice for all leiomyomas, however, some authors claim that for asymptomatic, non-obstructive and non-problematic leiomyomas follow-up may be sufficient since no malignant transformation of bladder leiomyoma has been reported. Reference Cornella J.L., Larson T.R., Lee R.A., et al.: Leiomyoma of the female urethra and bladder: Report of twenty-three patients and review of literature. Am J Obstet Gynecol, 1997, 176: 1278-1285.

Image-Dewaele et al.indd 263

Department of 1. Radiology, 2. Obstetrics and Gynecology, University Hospital Ghent, Ghent, Belgium.

5/08/14 14:39

JBR–BTR, 2014, 97: 264.

IMAGES IN CLINICAL RADIOLOGY Imaging findings in a patient with saliva in the auditory canal P. Gillardin1, P. Tomassen2, Ch. Vanclooster2, P. Vander Eecken2, M. Lemmerling1

A 78-year-old female presented with complaints of unilateral clear otorrhea, which worsened after consuming meals. The complaints started acutely four weeks before. There were no associated complaints of otalgia, craniofacial pain, hearing loss or tinnitus. During clinical examination, otomicroscopy revealed marked edema of the external ear canal with a limited amount of clear fluid and debris, and a normal tympanic membrane, as well as an anterior canal wall which was highly mobile during jaw movement. Pure tone audiometry showed bilateral symmetrical high frequency sensorineural hearing loss. Repetitive culture of the debris in the ear canal only detected commensal flora. Treatments with suction cleaning, several types of local antibiotics and local corticosteroids, and local application of caustic agents (lotagen) resulted in only temporary and partial improvement with multiple exacerbations over the course of six months. After resolution of edema of the external auditory canal, persistence of granulation tissue in the anterior bony canal wall a few millimeters lateral to the tympanic annulus became apparent, and led to the suspicion of a patent foramen tympanicum. Subsequent biochemical analysis of the ear discharge demonstrated an amylase concentration of more than 1500 U/l (normal values: undetectable). Cone-Beam CT examination was performed, which revealed a three-millimeter wide defect of the antero-inferior external auditory canal wall, six-millimeter lateral to the tympanic annulus (Fig. A, arrow). To detect a possible parotid fistula, MRI of the parotid region and petrous bone was performed. Analogous with the use of lemon-extracts in sialography to induce saliva production, patient was given a slice of lemon orally just before the examination. Axial high resolution T2 examination showed a small fluid effusion outlining the external auditory canal (Fig. B, arrows). Combined with the clinical investigation, patient was diagnosed with a patent foramen tympanicum and until now refused treatment.

Comment

The foramen tympanicum, also referred to as the foramen of Huschke, consists of an anatomical variation with an osseous defect in the antero-inferior wall of the external auditory canal, posteromedial from the temporomandibular joint. In a prospective study conducted by Lacout et al., 4.5% of the patients exhibited a foramen tympanicum. Presumably, an embryologic anomaly occurs during the formation of the external auditory canal, due to failure in fusion of the anterior and posterior prominences, resulting in a bony gap variant, the foramen of Huschke. As this osseous disparity may involute up to the age of 5, diagnosis should only be made after this age. In patients with chronic discharge, multiple mechanisms are possible including a parotid gland fistula or ectopic parotid tissue, lodged in the cleft. Synovial tissue of the temporomandibular joint may also herniate through this corridor. Caution should be warranted when executing temporomandibular arthroscopy in these patients, as the persistent foramen may lead to herniation of the endoscope in the external auditory canal and rupture of the tympanic membrane. An infection may thus spread in the external auditory canal, middle ear, as well as in the infratemporal fossa. Other complications include incudal dislocation, labyrinthine disruption or injury of the tympanic segment of the facial nerve. Patients with a foramen of Huscke also appear to be prone to tumoral or infectious spread from the infratemporal fossa to the external auditory canal and vice versa. Treatment is only performed in symptomatic patients, by way of patch placement obliterating the anomalous space.

Reference Lacout A., Marsot-Dupuch K., Smoker W.R., et al.: Foramen tympanicum, or foramen of Huschke: pathologic cases and anatomic CT study. AJNR Am J Neuroradiol, 2005, 26: 1317-1323.

Department of 1. Radiology, 2. Otolaryngology, General Hospital SintLucas, Ghent, Belgium.

Ima-Gillardin et al.indd 264

5/08/14 14:41

JBR–BTR, 2014, 97: 265.

IMAGES IN CLINICAL RADIOLOGY MR imaging features of acute bilateral caudate infarcts in pregnant woman M. Semnic1,2, R. Semnic2,3, K. Petrovic2,4, F.M. Vanhoenacker5,6,7

A 20-year-old female, in her 20th week of pregnancy, was hospitalized due to recurrent, severe vomiting, dehydration, somnolence and bilateral upper limbs athetosis. Metabolic status revealed ketonuria, proteinuria and hypokalemia. After urgent laparoscopy due to acute abdomen, the patient became hemodynamically unstable, with respiratory insufficiency and mechanical ventilation was applied. After stabilizing mental and clinical status, extubation procedure was performed. Neurological examination revealed psychomotor slowing, apathy, hyperreflexia and right sided rigor. Magnetic resonance imaging (MRI) of the brain revealed symmetric infarcts in head of both caudate nuclei. High signal intensity lesion on axial diffusion weighted image (DWI, b-1000 (Fig. A) in the head of both caudate nuclei and low signal on apparent diffusion coefficient image (Fig. B), in keeping with acute infarcts. Management of the patient included correction of fluid and potassium deficit and secondary stroke prevention with aspirin. Comment Acute bilateral caudate nucleus infarcts are rare and MRI findings in pregnant patient have not been described yet. Small vessel disease is the predominant cause followed by cardioembolic and underlying carotid disease, but presumed cause in this pregnant patient was the systemic hypoperfusion due to hypovolemic shock. The most prominent clinical features of caudate vascular lesions are behavioral and cognitive abnormalities. Bilateral caudate infarcts were previously attributed to frontal behavioral syndrome, often associated with significant memory impairment. Caudate nuclei damage in our patient caused consequent bilateral athetosis and disruption of fronto-subcortical circuits, resulting in psychomotor slowing and apathy. Due to bilateral involvement of caudate nuclei, development of “strategic-infarct dementia” could be expected. Reference 1. Tatemichi T.K., Desmond D.W., Prohovnik I.: Strategic infarcts in vascular dementia. Arzneim-Forsch/Drug Res, 1995, 45: 371-385.

Image-Semnic et al.indd 265

1. Neurology Clinic, Novi Sad, Serbia, 2. Faculty of Medicine, University Of Novi Sad, Serbia, 3. Institute of Oncology, Sremska Kamenica, Serbia, 4. Center of Radiology, Novi Sad, Serbia, 5. Department of Radiology, AZ Sint-Maarten Duffel-Mechelen, Mechelen, 6. Department of Radiology, Antwerp University Hospital, Edegem, Belgium, 7. University of Ghent, Faculty of Medicine and Health sciences, Ghent, Belgium.

5/08/14 14:43

JBR–BTR, 2014, 97: 266.

IMAGES IN CLINICAL RADIOLOGY Pseudo-achalasia: a complication of laparoscopic adjustable gastric banding P. Terryn1, J. Pringot1, L. Ghijselings1, B. Bomans2, P. Matthys1 A 49-year-old woman presented with dyspepsia and nocturnal regurgitation. A laparoscopic adjustable gastric binding (LAGB) had been performed 6 years before presentation. An upper gastrointestinal barium contrast study was performed and revealed a marked dilatation and tortuous course of the esophagus as well as absence of peristalsis and delayed evacuation of the esophagus (Fig. A, B). The findings were compatible with an achalasia-like disorder. An esophageal manometry revealed a constant high LES pressure with aperistalsis, thus confirming the diagnosis of (pseudo-)achalasia. Consequently a complete band deflation was conducted and resulted in a complete resolution of the patient’s symptoms. Two weeks later the control contrast study showed a marked improvement of the delayed evacuation and a small regain of peristaltic function. The dilatation and “sigmoidlike” image of the esophagus remained unchanged (Fig. C). Comment

Obesity is an important public health problem in all western countries with an increasing incidence and significant morbidity and mortality. LAGB is due to its limited invasiveness and therefore very low operative risk a frequent procedure for the management of obesity. It is however not free of complications and side effects with the most common being band erosion, band slippage, pouch dilatation, esophagitis and pseudo-achalasia. Pseudo-achalasia is in fact secondary achalasia, with a mechanical obstruction in the majority of the cases. This may include malignant disease, benign lesion or complications of surgical procedures at the distal esophagus or proximal stomach. The motility disorder is often indistinguishable from true achalasia through manometry or upper gastrointestinal barium contrast study. Approximately 3-4% cases of achalasia are in fact pseudo-achalasia. Pseudo-achalasia after LAGB is considered a rare complication (approximately 1,9%) but is generally underestimated with studies reporting up to 17%. Of all cases of pseudo-achalasia roughly 10% are caused by post-operative complications; with anti-reflux surgery being the most common. It is more frequent in patients with insufficiency of the LES (pre-existing or developed as a consequence of chronic pouch overfilling); furthermore it may occur secondary to band overinflation, band dislocation and placement of the band too far proximally at the level of the LES. The gold standard for the diagnosis of (pseudo-)achalasia is upper gastrointenstinal contrast study and esophageal manometry. The contrast studies are superior to computed tomography because of the ability to evaluate the esophageal motility. The role of CT is to rule out masses of or around the lower esophagus and to assist in the differentiation between pseudo-achalasia and achalasia. Upper gastrointestinal contrast studies must ensure correct band placement, rule out slippage or pouch dilatation as well as assess the motility and diameter of the esophagus. An esophageal diameter above 35 mm is considered as dilated. The degree of pathology can be further assessed through the radiological classification of Dargent with stage I being moderate dilatation with delayed emptying, stage II hypercontracting esophagus (tertiary waves) with poor emptying, stage III significant dilatation and stage IV for major achalasia-like dilatation or megaesophagus (our patient). The therapy consists of complete band deflation or removal of the band which often results in a significant decrease or complete resolution of symptoms due to the high reversibility of the disorder. Reference

1. Lipka S., Katz S.: Reversible pseudoachalasia in a patient with laparoscopic adjustable gastric banding. Gastroenterol Hepatol (N Y), 2013, 9: 469-471. 1. Department of Radiology, 2. Department of Surgery, Clinique Ste-Elisabeth Ziekenhuis, Brussels, Belgium.

image-terryn-.indd 266

5/08/14 14:44

JBR–BTR, 2014, 97: 267.

IMAGES IN CLINICAL RADIOLOGY A case of acute appendicitis at atypical localization I. Basara, M. Secil1

A 23-year-old male patient is admitted in the emergency unit with complaints of acute pain in the left lower quadrant of a bdomen, nausea, vomiting and anorexia. Physical examination, shows tenderness in the left lower quadrant, defense and rebound. Laboratory examination reveals a white blood cell count at 18,000 cells/mm3, with 73% neutrophils. His temperature is 38°c. The patient was referred to radiology unit and supine abdominal radiograph showed colonic gas distension in the left side of abdomen. Abdominal computed tomography (CT) examination showed that cecum was not at the normal localization in the left lower- middle part of abdomen. Similar to cecum, ascending colon was B also located in the left side of abdomen. The superior mesenteric vein (SMV) was located anterior and to the left of the superior mesenteric artery (Fig. A, thick arrow cecum, thin arrow SMV). At the inferior-medial part of cecum, a tubular, enhancing structure compatible with inflamed appendix was seen. The diameter and wall thickness of appendix were increased additionally there were mesenteric heterogeneity and linear density increases at periappendiceal region (Fig. B, thick arrow appendix, thin arrow periappendiceal h eterogeneity). The diameter of the appendix was 9 mm. In the light of these findings the patient was diagnosed as midgut malrotation and additionally left sided acute appendicitis. The patient was referred to general surgery department for surgery and discharged shortly after. Comment The most common surgical reason of abdominal pain is acute appendicitis. Sometimes acute appendicitis may settle in atypical localization. Left lower sided appendix may be with intestinal malrotation and situs inversus totalis (1, 2). In adults, intestinal malrotation is an asymptomatic situation compatible with abnormal fixation of midgut on peritoneal wall during the turning around superior mesenteric artery (1). The incidence of situs inversus varies from 1 in 6,000 to 1 in 35,000. Midgut malrotation is rarer than situs inversus. Even rarer is the occurrence of acute appendicitis associated with midgut malrotation. CT in our patient showed that the liver, stomach, and duodenum were located in their normal position. This finding showed that there is no presence of situs inversus. However, the cecum and ascending colon were located in the left side of the abdomen. The SMV was located anterior and to the left of the superior mesenteric artery, indicating midgut malrotation. Rarely as a result of malrotation, if midgut and cecum volvulus occurs, ischemia, obstruction or chronic abdominal pain may be seen. Other causes of acute abdominal pain are sigmoid diverticulitis, colitis, abdominal aortic aneurysm, renal colic, cystitis, epididymitis, prostatitis, testis torsion, incarcerated hernia, psoas abscess, tumor perforation, gynecologic reasons (ovarian tumors or cysts, adnexal torsion, ruptured ectopic pregnancy, pelvic inflammatory disease, endometriosis etc.) and acute appendicitis accompanying situs inversus (6). In the literature, it is reported that diverticular diseases may be misdiagnosed in acute appendicitis in the left sided cecum. Acute appendicitis with malrotation can be correctly diagnosed more than 90% success by CT examination. The CT findings of left-sided appendicitis are similar in appearance to those of right-sided appendicitis except for its opposite location. A tubular, enhancing structure surrounded by reactive changes in the adjacent fat suggests the diagnosis (2). Accurate preoperative diagnosis is critical in the management of acute appendicitis to prevent delay in the treatment of a relatively common disease, especially when it appears in an unusual location. CT not only provides accurate diagnosis of left-sided appendicitis but also is particularly useful in detecting associated rotational anomalies and related complications that may require separate surgical correction. References 1. Hou S.K., Chern C.H., How C.K., et al.: Diagnosis of appendicitis with left lower quadrant pain. J Chin Med Assoc, 2005, 68: 599603. 2. Kamiyama T., Fujiyoshi F., Hamada H., et al.: Left -sided acute appendicitis with intestinal malrotation. Radiation Medicine, 2005, 23: 125-127.

1. Department of Radiology, Dokuz Eylul University, Izmir, Turkey.

image-basara-.indd 267

5/08/14 14:45

JBR–BTR, 2014, 97: 268.

IMAGES IN CLINICAL RADIOLOGY Spontaneous osteonecrosis of the knee (SONK) A.M. Filip, S.B. Van den Broeck1

An 80-year-old woman presented at the emergency department with a sudden non-traumatic pain of the left knee. Supine decubitus plain radiographs of the left knee (Fig. A) reported no evidence of acute traumatic lesion, osteoarthritis bone remodeling, chondrocalcinosis nor mild joint effusion. Chondrocalcinosis crisis was subsequently considered as the final diagnosis and the patient received symptomatic treatment. Three months later, pain was still present and the patient underwent an MR exploration of the knee. T1-weighted (Fig. B) and proton density with fat saturation (PD FS) (Fig. C) sequences demonstrated a bone marrow edema of the whole medial femoral condyle, as well as a linear low signal intensity subchondral striation involving the weight-bearing portion of the medial condyle. The PD FS images showed evidence of collapse of the subchondral bone outside the striation, surrounded by a high signal synovial fluid rim. No reactive interface nor the typical “double line sign” of usual osteonecrosis were seen seen. Rupture of the posterior horn of medial meniscus with medial extrusion and also severe degenerative articular changes were present. These findings were consistent with spontaneous osteonecrosis of the knee. Given the wide extent of the lesion, total knee replacement surgery was planned. Comment

Spontaneous osteonecrosis of the knee (SONK) or “idiopathic osteonecrosis of the knee” was initially described as a distinct entity of osteonecrosis in 1968 by Ahlbäck, Bauer and Bohne. SONK mostly occurs in elderly individuals, generally after the seventh decade and women are commonly affected. The pain is of sudden onset and severe, causing significant functional disability. The lesion is generally unilateral and located in the load-bearing area of the medial femoral condyle which differentiates SONK from osteochondritis dissecans, preferentially localized in the lateral aspect of the medial femoral condyle and generally occurring in young patients. SONK differs from secondary avascular osteonecrosis (AON) which is always associated with risk factors such as alcoholism, corticosteroid therapy or systemic disease. At MR exploration, secondary AON differs from the idiopathic form with a typical “double line sign” that delineates the necrotic bone marrow, not present in SONK. In addition, at an early stage, SONK differs from secondary AON showing a curvilinear band of low signal in the sub-chondral bone, similar in appearance with stress fracture. C The etiology of SONK remains unknown. The acute onset of the symptoms, the clear predilection for elderly women often suffering from osteoporosis, the typical location in the load-bearing segment of the condyle and its similarities with stress fracture aspect on early MRI suggest that the initial lesion may be a repeated stress microfracture inducing local vascular impairment. This hypo thesis is reinforced by frequent association with medial meniscal tears or meniscectomy. It is considered that repeated and increased cartilage-to-cartilage or cartilage-to-meniscal fragment impact could damage subchondral bone. An early diagnosis of SONK can stop the evolution of the necrosis and avoid arthroplasty. In this view, MRI of the knee should be considered in case of non traumatic sudden pain of the knee in elderly women with non-contributing initial plain radiographs. Reference 1. Narváez J., Narváez J.A., Rodriguez-Moreno J., Roig-Escofet D.: Osteonecrosis of the knee: differences among idiopathic and secondary types. Rheumatology, 2000, 39: 982-989.

1. Department of Diagnostic Radiology, Clinique St-Luc, 5004 Bouge (Namur), Belgium.

image-filip-.indd 268

7/08/14 08:53

JBR–BTR, 2014, 97: 269.

LETTER TO THE EDITOR US diagnosis of acute pancreatitis caused by ruptured hydatid disease to the biliary system B. Karaman, B. Battal, S. Sari, S. Verim

Dear Sir, We read the recent article entitled “Ultrasonographic diagnosis of acute pancreatitis caused by ruptured hydatid disease to the biliary system” by Ozcaglayan O. et al. (1), published in JBR-BTR, with great interest. They have reported a case of acute pancreatitis, occurring by a hydatic cyst of the liver that ruptured into the biliary tract and describing the main disease and its complications by using ultrasonography (US), computed tomography (CT) and magnetic resonance imaging (MRI). They concluded that US is a very useful imaging modality for the diagnosis of hydatid disease and its complications, such as rupturing into the intrabiliary ducts and choledochus. It demonstrates hydatid material in their lumens and also provides differentiation from stones. In patients with hepatic hydatid cysts disease,

communications between the hydatid cyst cavity and the biliary tree can be either occult or frank (2). In our opinion new imaging modalities are needed for these communications. Because as the authors mentioned there can be complications like pancreatitis. Kantarci et al. reported that contrast enhanced magnetic resonance cholangiography (MRC) using gadolinium ethoxybenzyl diethylenetriamine pentaacetic acid (GdEOB-DTPA) and a 3D gradient echo technique is a robust tool that displays the biliary anatomy and provides functional information about physiological or pathological biliary flow (3). According to the present study, Gd-EOB-DTPA-enhanced MRC is the most sensitive and most accurate technique for the detection of biliary–cyst communication BCC in HHD. Therefore we want to suggest CE-MRC that can be added to T2weighted-MRC as a complementary

tool to increase the accuracy of BCC detection. References 1. Ozcaglayan O., Halefoglu A.M., Ozcaglayan T., Sumbul H.A.: Ultra sonographic diagnosis of acute pancreatitis caused by ruptured hydatid disease to the biliary system. JBRBTR, 2014, 97: 33-35. 2. Lee N.K., Kim S., Lee J.W., Lee S.H., Kang D.H., Kim G.H., Seo H.I.: Biliary MR imaging with Gd-EOB-DTPA and its clinical applications. RadioGraphics, 2009, 29: 1707-1724. 3. Kantarci M., Pirimoglu B., Ogul H., Bayraktutan U., Eren S., Aydinli B., Ozturk G., Karaca L.: Can biliary-cyst communication be predicted by GdEOB-DTPA-enhanced MR cholangio graphy before treatment for hepatic hydatid disease? Clin Radiol, 2014, 69: 52-58. doi: 10.1016/j. crad.2013.08.005. Epub 2013 Oct 22.

From: Gulhane Military Medical Academy, School of Medicine, Department of Radiology, Ankara, Turkey.

karaman-verim-(letter).indd 269

5/08/14 14:47

JBR–BTR, 2014, 97: 270.

ABSTRACT OF PAPER PRESENTED FOR FULL MEMBERSHIP AT THE BELGIAN SOCIETY OF RADIOLOGY BONE Conebeam-CT and fluoroscopy guided percutaneous absolute alcohol sclerotherapy of aneurysmal bone cysts – A single centre experience J. De Groote, T. Dewaele, L. Defreyne1 Purpose: Aneurysmal bone cysts are benign bone lesions representing approximately 1% of all bone tumors. Lesions usually manifest in the first or second decade of life and are most frequently eccentrically located in the metaphysis of long tubular bones or in the axial skeleton. Etiologically lesions are considered as benign intra-osseous vascular malformations but can become locally aggressive, with significant extraosseous expansion. Conventional treatment protocols consisted of surgical curettage, with or without associated bone grafting. In some cases, the localization and extent of the cyst are such that operative treatment is extremely hazardous. Moreover surgical curettage is associated with high recurrence rates varying from 10 to 44%. We present a less invasive treatment option with fluoroscopy and conebeam-CT guided direct percutaneous sclerotherapy with intralesional absolute alcohol injections. Material and methods: Fourteen patients diagnosed with an aneurysmal bone cyst, including 3 axial lesions and 11 lesions in the appendicular skeleton, were treated with direct percutaneous absolute alcohol (ethanol 96%) injections. Patients presented with symptoms of locoregional pain and/or disability including 2 cases with a perilesional pathologic fracture. Procedures were performed during general anesthesia. Preliminary fluoro-

scopic guided contrast injection and pre injection conebeam-CT ascertained correct needle placement. Intracystic contrast injection confirmed absence of significant venous drainage. The number of treatment sessions differed from one single session to four successive sessions. Maximum dose of ethanol injected during a single session did not exceed 0,5 ml/kg body weight. Three patients had a combined treatment protocol of percutaneous sclerotherapy and endovascular micro-embolisation of local afferent arterioles (with ethanol 96%). Seven patients had previous surgical curettage. Treatment response was evaluated clinically and with imaging using follow-up radiographs, conebeam-CT and/or MRI. Results: Twelve out of 14 patients showed significant clinical improvement post treatment with no residual local pain or disability nor future pathologic fractures. Average time of follow-up was 2 years and 1 month. Imaging response rates, evaluated in 9 patients with followup conventional radiography and/or conebeam-CT, showed signs of progressive bone formation at the location of the primary cystic lesions in 9 out of 9 patients. Follow-up MRI illustrated unchanged or reduced volume of the primary blood filled cystic lesions in 10 out of 11 patients with obliteration of typical fluid-fluid levels in 7 out of 11 patients. One patient had final surgical en-bloc resection following initial surgical curettage and 3 sessions of percutaneous sclerotherapy because of partial treatment response. One patient developed osteonecrosis of the talar corpus on follow-up MRI 3 months post treatment of a talar head lesion, treated conservatively with progressive weight bearing. Besides mild post procedural locoregional inflam-

matory symptoms, no other patients presented with significant peri- or postoperative complications. Conclusion: Conebeam-CT and fluoroscopy guided direct percutaneous absolute alcohol injection looks promising as a safe and effective treatment alternative to conventional surgical curettage of aneurysmal bone cysts. The minimal invasiveness of this technique makes successive treatment sessions possible in case of initial partial response or recurrent disease. Guidance with conebeamCT and fluoroscopy of low dose alcohol injections are rarely associated with local or systemic complications and form an effective technique to induce coagulative necrosis, efferent venous obliteration and secondary sclerosis in aneurysmal bone cysts. References 1. Lambot K., Pannier S., Grévent D., et al.: Primary aneurysmal bone cysts in children: percutaneous sclerotherapy with absolute alcohol and proposal of a vascular classification. Pediatr Radiol, 2012, 42: 599-605. 2. Cottalorda J., Bourelle S.: Current treatments of primary aneurysmal bone cysts. J Pediatr Orthop, 2006, 15: 155-167. 3. Cottalorda J., Bourelle S.: Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg, 2007, 127: 105-114. 4. Varshney M.K., Rastogi S., Khan S.A., Trikha V.: Is sclerotherapy better than intralesional excision for treating aneurysmal bone cysts? Clin Orthop Relat Res, 2010, 468: 1649-1659. 1. Department of Radiology, UZ Gent, Ghent, Belgium.

Advertising Index . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Pages GUERBET . . . . . . . . . . . . . . . . . . . . . . . . . . . IV BAYER . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . CIV

Contact details of advertising firms Bayer n.v./s.a.

Mommaertslaan 14 1831 Diegem Tel. 02/535.65.08 Fax 02/537.57.53 Mr. W. De Plecker

abstr full memb(bone).indd 270

Guerbet n.v./s.a.

Av. H. Dunantlaan 31 1140 Brussel Tel. 02/726 21 10 Fax 02/726 24 01 Mrs. N. Delavigne

14/08/14 14:02

PLATINUM SPONSOR

The Belgian radiology journal wishes to thank Olea medical for their continuous support

GOLD SPONSOR

Contrast for life The Belgian radiology journal wishes to thank Guerbet for their continuous support

BRONZE SPONSOR

The Belgian radiology journal wishes to thank Bracco for their continuous support

The Belgian radiology journal wishes to thank Toshiba for their continuous support

00b-JBR-Sponsoring kopie.indd 1

14/08/14 13:42

Integrated, Point of Care Solutions

Bayer NV/SA J.E.Mommaertslaan 14 B-1831 Diegem contact: fanny.goditiabois@bayer.com xxxxxxxxxxxxxxxxxxxxx