Official journal of the SocietĂ Italiana Specializzati in Chirurgia Odontostomatologica ed Orale Vol. 4 issue 1 April 2013 ISSN 2037-7525
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European journal of oral surgery Official journal of the SocietĂ Italiana Specializzati in Chirurgia Odontostomatologica ed Orale
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Vol. 4 issue 1 APRIL 2013
page 9
Keratocyst: literature review, case report and differential diagnosis with globulomaxillary cyst
page 13
Styloid-Stylohyoid Syndrome surgical treatment by intraoral approach
page 18
Conchal cartilage as interpositional graft material for the management of TMJ ankylosis
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II° SIMPOSIO OSTEOINTEGRATA MILANO DI
9.00-12.30
Carlo Maiorana
Comitato scientifico: Dario Andreoni Alfonso Baruffaldi Mario Beretta Marco Cicciù Sergio De Paoli Marco Finotti Giulio Rasperini Giano Ricci Piero Simeone Mariano Sanz Pascal Valentini
Si ringrazia:
HOTEL MARRIOTT
CORSI PRE-CONGRESSUALI
9.00-12.30
CORSO A: Alfonso Baruffaldi, Mario Beretta, Marco Cominetti, Alessandro Motroni Nuovi concetti nella progettazione e nel trattamento chirurgico-protesico mediante l’utilizzo di tecniche integrate di chirurgia guidata e cad/cam. CORSO B: Marco Finotti, Carlo Raimondo Protesi avvitata o cementata: l’utilizzo di diverse tecnologie e professionalità nel paziente implantoprotesico.
4–5
Ottobre 2013
PROGRAMMA VENERDÌ 4 OTTOBRE 14.00 14.15 plastica. 14.45
A PERTURA DEI LAVORI E SALUTO INAUGURALE Opening Lecture: Massimiliano Brambilla, specialista in chirurgia
Dall’estetica del sorriso all’armonia del volto: guida alla soddisfazione del narcisismo. Ia Sessione Scientifica Chairmen: Carlo Maiorana, Mario Beretta S. Marcus Beschnidt L’estetica in implantologia: stato dell’arte. 15.45 coffee break 16.15 Giulio Rasperini Il dente parodontalmente compromesso: conservazione o estrazione. 16.45 Gregorio Laino Cellule staminali: stato dell’arte e potenzialita’. 17.15 Pascal Valentini Short implants nella mandibola posteriore. 17.45 Discussione 18.00 Cocktail Entertainment
PROGRAMMA SABATO 5 OTTOBRE 9.00 10.30 11.00 12.30 13.00
IIa Sessione Scientifica Chairmen: Dario Andreoni, Piero Simeone Piero Venezia, Pasquale Lacasella La riabilitazione implantoprotesica nel paziente edentulo. coffe break Carlo Poggio L’occlusione nei pazienti riabilitati con protesi su impianti. Premiazione miglior poster. Presentazione caso clinico vincitore Premio Axel Kirsch 2012. Proclamazione vincitore Premio Axel Kirsch 2013. Chiusura lavori.
Con il patrocinio di:
Dipartimento di Scienze Clinico-Chirurgiche, Corso di Laurea Magistrale in Diagnostiche e Pediatriche Odontoiatria e Protesi Dentaria Sezione di Odontoiatria “S.Palazzi” Università degli Studi di Milano Università degli Studi di Pavia
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IMPLANTOLOGIA
European journal of oral surgery Official journal of the SocietĂ Italiana Specializzati in Chirurgia Odontostomatologica ed Orale
Editorial
Prof. Carlo Maiorana Editor-in-chief
Dear colleagues, I am very pleased to write you after the New Board of Siscoo was elected on last february. First of all, as new President of the Society I wish to thank, also on behalf of all the members, Prof. Franco Santoro: during his mandate as Director of the School of Specialty in Oral Surgery of Milan and President of the Society, he founded and grew up Siscoo, stimulating the new specialists to give their contribution in the European Journal of Oral Surgery, official organ of the Society. Within the next years we will work to give the better scientific and educational offer we can, waiting for the 2014 Congress. In the current year Siscoo has organized a course, on june the 22nd, focused on endodontic surgery. The course will be given by Dr Silvio Taschieri, a very well known specialist in the field. Next October, 4-5th, Siscoo is giving its patronage to the 2 Congress of the Camlog Italian Academy, in Milan. The two day congress will host foreign and italian speakers facing the hottest aspects in oral surgery , implantology and implant prosthodontics, giving also the participants the opportunity to attend pre congress courses. For all the Siscoo members first, but also for all the oral surgeons, this event will be a great opportunity to update their knowledge. The Board of the Society is also working to promote eJOS around the world, and we started receiving scientific contributions from abroad. I and the Editorial Manager of eJOS, Dr Federico Mandelli, strongly hope that each of you will contribute to spread eJOS in the country and around the world, by activating the personal contacts and submitting to the journal original clinical case reports and outcome of clinical trials . It doesnt’matter if you are not faculties, even the private practitioners aiming at excellence in their daily work are invited to bring their contribution. We have always been believing in the extraordinary power of synergies and hope that you all, even appreciating our efforts, will be on our side.
Dr. Federico Mandelli Editorial manager
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19/02/13 11:20
Review
Keratocyst: literature review, case report and differential diagnosis with globulomaxillary cyst Christian Pandini* Francesco Vettorello* Francesco Pallotti** Carlo Maiorana* Dental Clinic*, Department of Pathologic Anatomy** Fondazione IRCCS Policlinico CĂ Granda, University of Milan
Aim
Case report
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This was to underscore the primary importance of histological exams for an accurate differential diagnosis of cystic lesions, based not only on first and second level radiographs, considering the potential relapse of this lesion after its enucleation. The patient complained of a swelling in the right maxillary vestibular region between teeth 11 and 13; the orthopantomograph showed an inverted-drop shaped radiolucent area suggesting a globulomaxillary cyst; the CT Dental Scan confirmed the presence of vestibular resorption. Surgical excision was performed: the lesion appeared rounded and of approximately 1 cm in diameter. The histological examination revealed a cystic lesion covered by keratinized squamous epithelium (parakeratosis).
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Key words: Odontogenic keratocyst; differential diagnosis; histologic examination.
Pandini C. et al.
Introduction
in 1963, many clinicians reported different rates of recurrence, from 0% to 62% associated with various treatment options (13). One of these was curettage with a chemical solution (Carnoy’s) proposed by Stoelinga et al. The technique of decompression and marsupialization appeared in the scientific literature for more then 30 years. Furthermore, it has been noted that after decompression the cyst lining undergoes histologic changes resulting in its eventual replacement by oral epitelium (9). Some investigators found that the results of conservative management of OKCs are comparable to those of invasive surgery, as no differences in recurrence were observed. A clinical and radiografic follow-up is mandatory for years after surgery.
The odontogenic keratocyst (OKC) has been the object of great interest since Philipsen introduced the term in 1956 (1). A number of clinicians favor conservative therapy, while others prefer more aggressive forms of treatment. In the past decade, Nakamura proposed some conservative surgical approaches; lateral cystectomy, enucleation, cryosurgery, decompression, and marsupialization have been proposed in order to reduce the negative effects of an aggressive surgery and, thus, respecting the delicate anatomical structure of the jaws, giving the patient a better quality of life (1, 2). According to some authors like Batahine and Chow, OKCs have an incidence rate of about 12% to 14% of all odontogenic cysts with 2 peaks around the ages of 30 and 60 and seem to be more frequent in males (M/F 2:1). In 60% to 80% of the reported cases occur in the mandible, mainly found in the molar, angle, and ramus area. Usually a localized asymptomatic swelling is the most common symptom; spontaneous drainage of the cyst into the oral cavity and teeth mobility are also common. Nasal obstruction, paresthesia, and root erosion are more rare symptoms; some reports underline that OKCs can undergo malignant trasformation. OKC can be associated with an impacted tooth the cyst must be distinguished from a dentigerous cyst. Meiselman consider conservative therapies to include enucleation and marsupialization, Willliams et al. considering aggressive treatment a resetcion in addition to enucleation and resection whit or whitout loss jaw continuty. Keratocyst odontogenic tumor is a benign uni o multicystic intraosseus odontogenic tumor with the potential for local destruction especially when associated with Gorlin Goltz Syndrome (10). Toller and many clinicians infact consider enucleation and curettage a minimal reqiuirement, whereas the complete eradication is necessary in order to decrease or avoid recurrence (11, 12). Starting from the Pindborg and Hansen initial report
Case report A 61 years old male patient, LC, came to our observation complaining of a swelling in the front right maxillary region at the apex of teeth 11 and 13 (right maxillary central incisor and right upper canine). Clinical examination revealed the presence of a swelling of the vestibular region between the above mentioned teeth. Orthopantomographic examination revealed the presence of an inverted drop shaped radiolucent area, suggestive of a globulomaxillary cyst (Fig. 1). A CT Dental Scan was performed in order to have a second level radiographic analysis; it confirmed the presence of a wide area of vestibular resorption as well as the partial absence of the cortical plate (Fig. 2, 3). Teeth 11 and 13 gave a positive response to the vitality test. Enucleation of the lesion was decided. After giving local anaesthesia with articaine 1:100000 and buccal and palatal infiltration, a mucoperiosteal trapezoidal flap was prepared with distal and mesial releasing incisions (Fig. 4, 5). After enucleation, the lesion appeared rounded and of approximately 1 cm in diameter (Fig. 7), was preserved
FIG. 1 Orthopantomography showing the lesion between the roots of teeth 13-11: it has the typical inverted drop shape.
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FIG. 2 CT Dental Scan, panoramic section.
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Review
FIG. 3 CT, orthoradial section.
FIG. 4 Intraoral front view showing a small tumescence.
FIG. 5 Intraoperative view of the mucoperiosteal flap.
FIG. 6 The vestibular cavity after enucleation shows vestibular cortical bone resorption.
FIG. 7 The lesion measured.
FIG. 8 10x magnification, showing the formation of cellular debris. FIG. 9 Detail at 40x magnification.
in 10% buffered formalin and then sent to the Institute of pathological anatomy for histological examination, which revealed a cystic lesion covered by keratinized squamous epithelium (parakeratosis) with lumen full of horny lamellas and cellular debris. Slides were stained with haematoxylin and eosin. The 40x magnification highlighted a cystic lesion showing parakeratosis (Fig. 9).
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Pandini C. et al. Histology revealed a keratocystic odontogenic tumor. Since the literature reports a high tendency to relapse in the 2 to 10 years after enucleation, the follow-up is scheduled every 6 months for the first two years and then for an indefinite time to check for possibile relapse.
Conclusion This case report shows the primary importance of a histological examination in order to distinguish the different types of cystic lesions, without having to use first or second level X-rays as the only validating exam for diagnostic purposes. In fact, in the described case, clinical signs and x-rays revealed a cyst in the front region which was clinically differentiated as “odontogenous cyst” from a “non-odontogenous cyst”, whereas the histologic analysis revealed a keratocystic odontogenous tumour. To this end the patient will undergo a periodical followup in order to detect the relapse of the lesion or its transformation into a different lesion with a more severe prognosis.
References 1. Marker P, Brøndum N, Clausen PP, Bastian HL. Treatment of large odontogenic keratocysts by decompression and later cystectomy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1996 Aug;82(2):122-31. 2. Nakamura N, Mitsuyasu T, Mitsuyasu Y, Taketomi T, Higuchi Y, Ohishi M. Marsupialization for odontogenic keratocysts: Long-term follow-up analysis of the effects and changes in growth characteristics. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002 Nov;94(5):543-53. 3. Bataineh AB, Al Qudah MA. Treatment of mandibular odontogenic keratocysts. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1998 Jul;86(1):42-7. 4. Chow HT. Odontogenic keratocyst: A clinical experience in Singapore. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1998 Nov;86(5):573-7. 5. Schmidt BL, Pogrel MA: The use of enucleation and liquid nitrogen cryotherapy in the management of odontogenic keratocysts. J Oral Maxillofac Surg. 2001 Jul;59(7):720-5. 6. Zhao YF, Wei JX, Wang SP. Treatment of odontogenic keratocys: A follow-up of 225 Chinese patients. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002 Aug;94(2):151-6. 7. Yoshida H, Onizawa K, Yusa H: Squamous cell carcinoma arising in associaton with an orthokeratinized odontogenic keratocyst. Report of a case. J Oral Maxillofac Surg. 1996 May;54(5):647-51. 8. Pitak-Arnnop P, Dhanuthai K, Hemprich A, Pausch NC. Follicular cysts, odontogenic keratocysts, and Gorlin-Goltz syndrome: some clinicopathologic aspects. J Craniofac Surg. 2011 May;22(3):1170. 9. Giuliani M, Grossi GB, Lajolo C, Bisceglia M, Herb KE. Conservative management of a large odontogenic keratocyst: report of a case and review of the literature. J Oral Maxillofac Surg. 2006 Feb;64(2):308-16. 10. Borgonovo AE, Di Lascia S, Grossi G, Maiorana C. Two-stage treatment protocol of keratocystic odontogenic tumour in young patients with Gorlin-Goltz syndrome: marsupialization and later enucleation with peripheral ostectomy. A 5-year-follow-up experience. Int J Pediatr Otorhinolaryngol. 2011 Dec;75(12):1565-71. 11. Toller P. Origin and growth of cysts of the jaws. Ann R Coll Surg Engl. 1967 May; 40(5): 306–336. 12. Philipsen HP. Om keratocyster (kolestetomer) i kaeberne. Tandlaegebledet. 1956;60:963–981. 13. Voorsmit RACA, Stoelinga PJW, van Haelst UJGM. The management of keratocysts. J Maxillofac Surg 1981;9:228-236.
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Case report
Styloid-Stylohyoid Syndrome surgical treatment by intraoral approach Felipe Ladeira Pereira* Meire Yurie Sonohara** Antônio José Araújo Pereira Junior*** Liogi Iwaki Filho**** Ângelo José Pavan**** Edevaldo Tadeu Camarini**** *DDS. Oral and Maxillofacial Surgeon. Brazilian Army. Juiz de Fora General Hospital (HGeJF), Juiz de Fora, Minas Gerais, Brazil. **DDS. Private Practice, Maringá, Paraná, Brazil. ***DDS, MS student. Oral and Maxillofacial Surgeon. Andaraí General Hospital, Rio de Janeiro, Rio de Janeiro, Brazil. ****DDS, PhD. Professor of Oral and Maxillofacial Surgery. Maringá State University (UEM), Maringá, Paraná, Brazil.
Background
Case report
Conclusion
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The obscure symptoms that normally accompain the Styloid-Stylohyoid Syndrome may lead the patient to seek different specialists before a final diagnosis is achieved. Diagnosis of Styloid-Stylohyoid Syndeome is very difficult both for the extensive symptoms and for the regional anatomy of the styloid process, its proximity to large vessels of the neck and to nerves of the face, such as the glossopharyngeal, vagus, accessory and hypoglossal. Patient R.C.G.A., a 31-years-old Caucasian female, sought the Oral Pathology Ambulatory from the Maringá State University complaining of pain during neck movement and foreign body sensation in the right side. Clinical examination pointed to Styloid-Stylohyoid Syndrome. The radiographic examination disclosed ossification of the stylohyoid ligament confirming the diagnostic hypothesis. The patient was submitted to surgical procedure under general anesthesia and the process was removed by an intraoral approach. At postoperative evaluation, there was total remission of symptoms and the patient is still under follow-up after 8 years. The purpose of this paper is, in addition to reporting the case, to discuss the advantages and disadvantages of intra and extraoral approaches by means of a short literature review, as well as the significance of imaging in the selection of the ideal surgical approach to this disease.
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Key words: Styloid Process; Eagle’s Syndrome; Oral Surgery; Intraoral Approach.
Felipe Ladeira Pereira et al.
Introduction The first reports on enlargement of the styloid process date from the 17th century when anatomists explored this region. These post mortem findings were only useful as a mere anatomical curiosity and had no clinical correlation. In 1937 W.W. Eagle reported various cases of a cervicopharyngeal symptomatology and associated them with radiographic findings. It was believed that trauma in the cervicopharyngeal region, especially after tonsillectomy, might stimulate a subsequent growth of the styloid process (3-5, 8, 11). Various names were proposed for the syndrome: Styloid Process Neuralgia (7), Styloid Syndrome (7, 11, 21), Stylohyoid Syndrome (7, 11), Elongated Styloid Process Syndrome (22, 24)Eagle’s Syndrome (2, 4, 5, 7, 11, 19, 20, 23) and Styloalgia (15, 18). The most appropriate, however, is Styloid-Stylohyoid Syndrome since such anomalies may be of the styloid process, of the stylohyoid ligament or a combination of both (11), although it has been divided into four in recent literature (4). Differential diagnoses are innumerous because many of the symptoms detected in the enlargement of the styloid process (orofacial pains and dysfunctions) are also found in patients without presence of the elongated process. Furthermore, its attachments may be susceptible to stretch and whiplash type injuries in which an acute force exceeds the physiological limits of the temporal bone attachment (14, 16). The objective of this paper is to present a case report of stylohyoid ligament ossification removed by intraoral approach under general anesthesia.
Case report R.C.G.A., 31 years old, Caucasian female, sought the Oral Pathology Ambulatory from the Dental Department of the Maringá State University complaining of foreign body sensation in her throat and pain while turning her head. The patient could locate with her forefinger a slender projection in the posterior region of her right mouth floor.
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Palpation of the elongated styloid process produced exacerbation of the symptoms, as did the palpation of the right retromandibular region and the medial pterygoid muscle. Local anesthetic infiltration in the right tonsillar fossa was performed, with 1 cartridge of 2% mepivacaine. The patient felt total remission of symptoms after a few minutes, confirming the hypothesis of StyloidStylohyoid Syndrome. She denied prior cervical trauma or tonsillectomies. Panoramic x-ray (fig. 1) and coronal Computed Tomography (CT) (fig. 2) were asked, revealing an ossified stylohyoid ligament with two pseudoarticulations in the right mandibular angle region. The contralateral side also showed an abnormal length styloid process, but the patient had no complains on her left side. The process was removed by intraoral approach, since it was easily palpable in the right palatoglossus muscle region. General anesthesia via nasotracheal intubation was conducted, and a 2 cm incision performed over the mucosa that lied upon the most prominent tip of the process, medially to the lingual nerve. After proper muscular retraction, the overlying periosteum was incised and stripped from the process (fig. 3). A curved Kelly clamp was placed 3 mm above the distal pseudoarticulation, retracting the soft tissues and allowing clear visualization of the exposed process. This pseudoarticulation was disarticulated and the distal fragment of the elongated styloid process removed (fig. 4). The remaining process left was within the normal limits, as mentioned by Chase et al. (1986) (5) and Yavuz et al. (2011) (24), and the removed segment measured 2.2 cm (fig. 5). Mucosa suture was performed as usual. Antibiotics, non-steroid antiinflammatories and analgesics were prescribed in the postoperative period. The patient still continues asymptomatic after 101 months (more than 8 years) of follow-up (Fig. 6).
Discussion The styloid process is a slender projection connected to the inferior aspect of the petrous part of the temporal bone just below the tympanic
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membrane and behind the tympanic plaque which shields its attachments. It lies behind the pharyngeal wall of the palatine fossa, between the internal and external carotid arteries (16). Innervation comprises the glossopharyngeal nerve in the posterior lateral wall of the tonsillar fossa (medial to the process), the facial nerve emerging from the stylomastoid foramen which is slightly posterolateral to the base of the styloid process. The accessory nerve, the hypoglossal nerve and the vagus nerve are placed medially to the process, together with the internal jugular vein and the internal carotid artery with its sympathetic chain (14, 16). The normal length of the styloid process has many values in the pertinent literature, varying among 1.52 cm (15) to 4.77 cm (15), with a mean of 2.5 cm (9, 11-13, 24). Steinmann (1968) (21) proposed three theories to explain such ossification. The “Theory of Reactive Hyperplasia” implies that if the styloid process is adequately stimulated as in pharyngeal trauma, ossification would take place in the terminal portion of the process at the expense of the stylohyoid ligament. The “Theory of Reactive Metaplasia” also involves traumatic stimulus which would induce some sections of the stylohyoid apparatus to undergo metaplastic changes and thereby become intermittently ossified. Finally, the “Theory of Anatomical Variance” involves the stylohyoid ligament and/or the styloid process as ossified structures that develop in the early, formative years after birth. This theory may fit in those cases in which there are early radiographic findings of such ossification in children and young adolescents in the absence of antecedent cervicopharyngeal trauma (as an inductive stimulus) (4). This could not be adjusted to the Classical Eagle Syndrome because there is no prior trauma (9). Camarda et al. (1989) (4) added the “Theory of Aging Developmental Anomaly”, in which there is an increased inelasticity of the soft tissue. This may lead to the development of tendinosis in the junction of the stylohyoid ligament with the lesser cornu of the hyoid bone, secondary to the increased ligament resistance to joint movement (between ligament and bone) in some older patients (4). As such, there are four syndromes: the Classical Eagle Syndrome and
Case report
fig. 1 Panoramic x-ray showing the ossified ligament with its two pseudoarticulations.
fig. 2 Coronal CT showing the total extent of the ossified stylohyoid ligament and the proximity of its tip to the oral mucosa.
fig. 4 Surgical site after disarticulation of the distal process fragment.
fig. 3 Process tip after the periosteum stripping.
fig. 5 Removed process.
the Carotid Artery Syndrome, which reactive hyperplasia and metaplasia ossification theories explains the first and maybe the second (once trauma is not necessarily the cause of the Carotid Artery Syndrome); the Stylohyoid Syndrome which would be justified by the theory of the anatomical variation, and finally the Pseudostylohyoid Syndrome explained by the theory of aging developmental anomaly (4). Although ossification of the stylohyoid
fig. 6 101 months postoperative panoramic x-ray.
ligament complex is fairly common, the abnormality rarely causes symptoms. Of the 2% to 4% of the general population who presents radiographic evidence, the majority are asymptomatic (2, 4, 6, 7, 9-11, 14, 18, 19, 24, 25). The symptoms of the Classical Eagle Syndrome and the Carotid Artery Syndrome are well described in literature (1-4, 6, 7, 11, 13, 14, 16, 18, 19, 21-24). The symptoms of the other two are the same of the Classic, but without prior
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trauma history and involving a specific age group, usually above 40 years for the Pseudostylohyoid Syndrome and not necessarily for the Stylohyoid Syndrome (11). In the Classical Eagle Syndrome, the chief complaint is a continuous throat pain during convalescent period, in patients submitted to tonsillectomy, and a sensation of foreign body lodged in the throat (2, 5, 7, 11, 1820, 23, 24) and during functional movements, such as eating, yawning, vol. 4 n. 1 2013
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Felipe Ladeira Pereira et al. turning the head and swallowing (16, 17, 19). Pain is related to swallowing and speech, being frequently referred to the ear on the side of the elongated styloid process (2, 5, 11, 12, 16, 17, 19, 23). It is assumed that healing tonsillectomy scar tissue tightens the mucosa across the tip of the elongated styloid process and that movements of this mucosa during function across it is thought to cause the symptoms (5, 7, 16, 19, 23, 24). Pharyngeal pains are theoretically generated by stretching or fibrous compression of the V, VIII, IX and X cranial nerve endings in the tonsillar fossa during the healing phase (5, 7, 11, 23). When the stylohyoid ligament is ossified, contraction of the stylopharyngeal muscle lifts the pharynx upwards and laterally and, with the ossified ligament remaining fixed in this maneuver, the glossopharyngeal nerve is pulled across it during the swallowing act and may be stimulated mechanically to produce pain (11). An important feature of Carotid Artery Syndrome is that it is not dependent on a tonsillectomy. Because the styloid process lies between the internal and external carotid, any deviation in the process or ossification of the ligament may produce pressure on either of these structures and produce regional carotidynia (pericarotid sympathetic plexus irritation). When pressure is exerted on the external carotid artery, the pain is regionalized to the anatomic structures supplied by the artery. Clinically, the patient may complain of constant pain in the neck, pain on turning the head, regional carotidynia, or tenderness of a cervical lymphnode. Pressure on the internal carotid artery may produce symptoms as a result of the unique arterial supply to the cranial cavity and its contents. Symptoms of carotid artery syndrome present as chronic neck pain, pain on turning the head, and pain irradiating to the eye (3, 7, 10, 11, 13, 16, 17, 19, 23, 24). Normally, panoramic radiography is indicated for best visualization of the styloid process (1, 11, 13, 18, 19), but other x-ray options such as the anteroposterior (3, 11, 19), lateral cefalometric (3, 11, 13, 19) and Towne’s view (2, 24) help to visualize the process in two planes. The use of two-dimensional and currently tridimensional CT is better for defining length, angulation and anatomic relationships of the styloid process (2, 5, 10, 12, 20).
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Regarding prevalence of gender, the subject is quite controversial. Some authors declare that the incidence is equal for both genders (6, 8, 10, 19), while others that females have a greater tendency to develop this pathology (1, 8, 10, 12, 18, 21, 24). The mean age also varied in the literature, ranging from among 4 to 94 years in patients presenting radiographic abnormalities (4, 6, 10, 11, 18, 22, 24), usually symptomatic over 30 years old (2, 4-6, 7, 10-12, 14, 18, 23-25). There are many differential diagnoses concerning the oral and maxillofacial area and other ones and they are presented in Table I. In physical examination, tonsillar fossa palpation along the occlusal plane is performed, where it is possible to feel the tip of the process, which exacerbates the symptoms and local sensitivity (the process cannot be felt while in its normal size) (1-3, 5, 11, 12, 17-19, 23-25). After a few minutes of the infiltration of local anesthetic at the site at which the styloid process was palpable on the tonsillar fossa, the patient´s symptoms and local tenderness temporarily subsides and the result of the test is regarded as positive (2, 11, 18, 19, 23). Nevertheless, to Yavuz et al. (2011) (24), anti-inflammatory drug therapy were prescribed preoperatively to rule out tendinitis or myositis, but none of them were relieved from their complaints after medication. In the past, the manual fracture of the elongated styloid process was advocated, but postoperative results were unsatisfactory (2, 3, 5, 7, 10, 11). Surgery can be performed under local (2, 5, 7, 22, 25) or general anesthesia (2, 7, 11, 17, 18, 20, 24). Local anesthesia allows a quicker surgery and rapid postoperative recovery (5, 22, 25). Morbidity and mortality associated with general anesthesia are reduced (5, 22, 25). In our department, we believe that surgery should be performed under general anesthesia in order to guarantee more comfort to the patient and better control of possible complications. It is accepted that intraoral approach should only be done if it is possible to palpate the process in the tonsillar fossa (3, 5, 7, 25), as happened in the reported case. In the presence of palatine tonsils, tonsillectomy should be performed first (10, 18, 22, 24, 25) and special care is necessary not to injure the closely associated structures
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(external and internal carotid arteries and the glossopharyngeal nerve) during detachment (11). The authors that advocate the intraoral approach believe that it is safe and relatively easy while avoiding cutaneous scars and extensive dissections (2, 7, 10, 14, 18, 20, 22, 24, 25). Its drawbacks are infection possibility of deep cervical spaces, poor visualization of the surgical field, increased risk of vascular (and its subsequent control) and nervous damage (VII and VIII) (2, 3, 7, 10, 12, 14, 17, 18, 22-25). The extraoral approach advantages are: better visualization and exposure of the process and its associated structures (2, 3, 7, 10, 18, 20, 24, 25), especially of the bifurcation of the external carotid with the maxillary artery and the superficial temporal artery (3, 23, 25); if hemorrhage of a larger vessel takes place, it can be handled in a controlled manner and with good vision (2, 3, 7, 10, 20); the use of a sterile surgical approach reduces the risk of bacterial contamination (2, 3, 7, 10, 18, 24, 25) and finally, this approach further permits a more ample resection of the styloid process (5, 23). The disadvantages are: larger dissection and surgical intervention (drains, sutures) (5, 10, 20, 24, 25), presence of a skin scar (2, 3, 7, 10, 18, 20, 24, 25), the need of general anesthesia (2, 7, 25), longer recovery and a possibility of impairing the facial nerve (2, 3, 14, 18). Tiago et al. (2002) (23) reported that the scar is aesthetically acceptable. More recent works report the use of anesthetic associated with corticosteroids in periodical inoculations in the site, based upon the presence of an inflammation, but long term results has not been substantiated (2, 3, 5, 7, 10, 16, 18, 20, 24). This, combined with an intraoral appliance that restricts eccentric mandibular movement during the healing phase may be responsible for significant long reduction in pain and dysfunction (16). The current literature tendency is to name the syndrome after the ossification theory that better explains it (17). Since the patient denied prior cervical trauma and was 31 years old at the time of the surgery, she could be classified in the description of the Stylohyoid Syndrome.
Case report
tab. 1 Differential diagnosis of the Styloid-Stylohyoid Syndrome.
Conclusion The Styloid-Stylohyoid Syndrome is usually difficult to diagnose, since its symptoms are similar to those of other pathologies that involve adjacent tissues and structures of the oral and maxillofacial region (such as neural, vascular, muscular and articular). Treatment options must be carefully evaluated for each case. Radiographic examination of good quality is paramount for an adequate planning and, when indicated, the surgical approach is one of the best ways of treatment, since our patient is still asymptomatic after more than 8 years.
References 1. Aral IL, Karaca I, Güngör N. Eagle’s syndrome masquerading as pain of dental origin. Case report. Aust Dent J. 1997;42:18-9. 2. Beder E, Ozgurzoy OB, Ozgurzoy SK. Current diagnosis and transoral surgical treatment of Eagle’s syndrome. J Oral Maxillofac Surg. 2005;63:1742-5. 3. Breault MR. Eagle’s syndrome: review of the literature and implications in craniomandibular disorders. Cranio. 1986;4:324-37. 4. Camarda AJ, Deschamps C, Forest D. Stilohyoid chain ossification: A discussion of etiology. Oral Surg Oral
Med Oral Pathol. 1989;67:508-14. 5. Chase DC, Zarmen A, Bigelow WC, McCoy JM. Eagle’s syndrome: a comparison of intraoral versus extraoral surgical approaches. Oral Surg Oral Med Oral Pathol. 1986;62:625-9. 6. Correl RW, Jensen JL, Taylor JB, Rhyne RR. Mineralization of the styloid-stylomandibular ligament complex. Oral Surg Oral Med Oral Pathol. 1979;48:28691. 7. Diamond LS, Cottrell DA, Hunter MJ, Papageorge M. Eagle’s syndrome: a report of 4 patients treated using a modified extraoral approach. J Oral Maxillofac Surg. 2001;59:1420-5. 8. Eagle WW. Elongated styloid processes: report of two cases. Arch Otolaryngol. 1937;25:584-7. 9. Eagle WW. Symptomatic elongated styloid process: report of two cases of styloid process-carotid artery syndrome with operation. Arch Otolaryngol. 1949;49:490-503. 10. Fini G, Gasparini G, Filippini F, Becelli R, Marcotullio D. The long styloid process syndrome or Eagle’s syndrome. J Craniomaxillofac Surg. 2000;28:123-7. 11. Gossman Jr JR, Tarsitano JJ. The styloid-stylohyoid syndrome. J Oral Surg. 1977;35:555-60. 12. Hossein R, Kambiz M, Mohammad D, Mina N. Complete recovery after an intraoral approach for Eagle syndrome. J Craniofac Surg. 2010;21:275-6. 13. Langlais RP, Miles DA, Van Dis ML. Elongated and mineralized stylohyoid ligament complex: a proposed classification and report of a case of Eagle’s syndrome. Oral Surg Oral Med Oral Pathol. 1986;61:527-32. 14. Miller DB. Eagle’s syndrome and the trauma patient. Funct Orthod. 1997;14:30-5. 15. Moffat DA, Ramsden RT, Shaw HJ. The styloid syndrome: aetiological factors and surgical management. J Laryngol Otol. 1977;91:279-94.
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16. Palesy P, Murray GM, Boever J, Klineberg I. The involvement of the styloid process in head and neck pain - a preliminary study. J Oral Rehabil. 2000;27:275-87. 17. Pereira FL, Filho LI, Pavan AJ, Farah GJ, Goncalves EA, Veltrini VC, et al. Styloid-stylohyoid syndrome: literature review and case report. J Oral Maxillofac Surg. 2007;65:1346-53. 18. Prasad KC, Kamath MP, Reddy KJM, Raju K, Agarwal S. Elongated styloid process (Eagle’s syndrome): a clinical study. J Oral Maxillofac Surg. 2002;60:171-5. 19. Sivers JE, Johnson GK. Diagnosis of Eagle’s syndrome. Oral Surg Oral Med Oral Pathol. 1985;59:575-7. 20. de Souza Carvalho AC, Magro Filho O, Garcia IR Jr, de Holanda ME, de Menezes JM Jr. Intraoral approach for surgical treatment of Eagle syndrome. Br J Oral Maxillofac Surg. 2009;47:153-4. 21. Steinmann EP. Styloid syndrome in absence of an elongated process. Acta Otolaryngol. 1968;66:347-56. 22. Strauss M, Zohar Y, Laurian N. Elongated styloid process syndrome: intraoral versus extraoral approach for styloid surgery. Laryngoscope. 1985;95:976-9. 23. Tiago RSL, Marques Filho MF, Maia CAS, Santos OFS. Sindrome de Eagle: avaliação do tratamento cirúrgico. Rev Bras Otorrinolaringol. 2002;68:196-201. 24. Yavuz H, Caylakli F, Erkan AN, Ozluoglu LN. Modified intraoral approach for removal of an elongated styloid process. J Otolaryngol Head Neck Surg. 2011;40:8690. 25. Zohar Y, Strauss M, Laurian N. Elongated styloid process syndrome masquerading as pain of dental origin. J Maxillofac Surg. 1986;14:294-7.
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Case report
Conchal cartilage as interpositional graft material for the management of TMJ ankylosis Anuj S Dadhich* Kumar Nilesh* Pranav Asher** *M.D.S. (Oral & Maxillofacial Surgery) Lecturer MGV Dental College & Hospital, Nashik, India **M.D.S. (Oral & Maxillofacial Surgery) Reader, School of Dental Sciences, KIMSDU, Karad, India.
Aim
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Ankylosis may be defined as the fusion of the articular surfaces with bony or fibrous tissue. Temporomandibular Joint (TMJ) ankylosis is one of the most debilitating condition affecting the facial skeleton and its surgical correction is limited by a high recurrence rate, particularly in patients undergoing surgery without the use of interpositional materials. A variety of interpositional materials, both alloplastic ones and autografts, and alloplast have been used to prevent recurrence after arthroplasty. This article reports on the use of conchal cartilage as interpositional material in the surgical treatment of TMJ ankylosis in a young female patient with unilateral TMJ ankylosis.
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Key words: Temporomandibular joint; Ankylosis; Conchal cartilage.
Case report
Introduction Ankylosis of TMJ is one of the most debilitating conditions affecting the facial skeleton. Recurrence is a significant postoperative complication after release of TMJ ankylosis. A relapse rate as high as 50% has been reported (1). In order to prevent or minimize such relapse, the use of various allografts and autografts as interpositional material has been advocated. Although the use of alloplasts eliminates the need for a donor area, their use has been associated with various complications like implant fragmentation, migration, foreign body reaction etc. (2-4). In comparison autogenous grafts are more popular as interpositional material. A variety of autogenous materials have been used, such as costochondral grafts, dermis, and temporalis muscle or fascia. Many investigators believe that the choice of interposition material is important in
fig. 1 Preoperative extraoral view: mouth opening is about 4 mm.
preventing recurrence (5, 6). This article reports and discusses the advantages of the use of free conchal cartilage as interpositional graft material for the management of TMJ ankylosis.
Case report A 19 years old female patient was referred to the Oral & Maxillofacial surgery clinic with chief complaint of restricted mouth opening from childhood. From the collection of a detailed case history it was found that she had had a trauma to the chin region when she was about the age of 2 years, for which no treatment was taken. During the examination the mouth opening was only around 4 mm (fig. 1). Extra-oral examination revealed facial asymmetry with fullness on right side and deviation of chin on the right side. On palpation the movements of TMJ were not felt
on the right side. A cone beam CT was done which showed complete bony union between the ramus of the mandible and the glenoid fossa (fig. 2). Release of ankylosis was planned by interpositional arthroplasty under GA. The patient was intubated by fiber optic intubation technique using a 4mm diameter flexible bronchoscope (Storz) and a no. 7 armored tube. The TMJ was exposed using a standard preauricular incision, after incising skin, superficial fascia and temporoparietal fascia was incised to expose the ankylotic mass. Multiple burr holes were drilled and later were joined to complete the gap arthroplasty. A gap of appox. 1.5 cm was created between the ramal segment and glenoid fossa. The conchal cartilage approx. 2 x 1 cm was harvested via an incision in posterior region of the helix (fig. 3). The cartilage was shaped and sutured to the residual disc and also to the holes drilled in the bone, so as to form a barrier between the two segments, in order to prevent
fig. 2 Preoperative Cone Beam Tomogram: a) 3D CT showing complete bony union between the ramus and the glenoid fossa; b) axial view taken at level of TMJ showing complete obliteration of join space with bony union between condyle-ramus and gleniod fossa (block arrow).
fig. 4 Conchal cartilage sutured in place as interpositional graft. fig. 3 A: incision for harvesting the conchal cartilage. B: the harvested cartilage.
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fig. 5 Postoperative mouth opening at 18 months (about 32 mm).
development of reankylosis (fig. 4). The patient underwent extensive physiotherapy in the immediate postoperative period. At the 18 month post-operative follow-up the patient retained a mouth opening of about 32 mm and had no other significant complaint (fig. 5). The donor site also showed no residual defect. The postoperative radiograph showed a pseudo-joint on the right side without any bony mass or growth between the newly created joint surfaces.
Discussion There he treatment of ankylosis varies from condylectomy, gap arthroplasty to interpositional arthroplasty with or without costochondral grafting. There is a variety of materials used as interpositional material, which are either autogenous or alloplastic materials; their main goal being to form a physical barrier between the raw bony surfaces of the ramus and the glenoid fossa which are formed through the osteotomy procedure carried out during gap arthroplasty. The temporalis myofascial flap is very frequently used for this purpose, however it is associated with complications such as temporal hollowing and the procedure of harvesting the temporalis myofascial flap is difficult and lengthy, prolonging the operating time. The use of conchal cartilage as autograft harvested from various parts of the body has been successfully
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reported owing to the low metabolism characteristic the of cartilage, so that no foreign body reactions usually occur. Tucker et al (8) reported that in a comparative study in monkeys the use of auricular cartilage as interpositional material after dissection and high condylar shave, the cartilage trophism was not altered and degenerative alterations of the joint occurred in less cases in the test group than in controls, in which the disc was removed and no interpositional graft was placed. Lei (7) reported successful clinical use of conchal cartilage after TMJ ankylosis in seven pediatric patients. The usefulness of conchal cartilage in TMJ arthroplasty include: a) readily available autogenous tissue; b) the graft is relatively easy and quick to harvest; c) it can be harvested from the same operative field; d) the graft remains inert and does not undergo resorption for a long period of time; thus it acts as an excellent barrier to separate the ramal stump from the glenoid fossa; e) it does not cause any aesthetic deformity of the donor site; f) the contour of the cartilage fits the condyle process well. The final outcome of the case reported in this article supports the use of autologous conchal cartilage as interposition autograft as a successful treatment option for TMJ ankylosis.
References 1. Topazian RG. Comparison of gap and interposition arthroplasty in the treatment of temporomandibular joint ankylosis. J Oral Surg 1966;24:405. 2. Dolwick MF, Aufdemorte TB. Sillione-dnduced foreign body reaction and lymphadenopathy after temporomandibular joint arthroplasty. Oral Surg 1983;59:449. 3. Fontenot MG, Kent JN. In vitro wear performance of Proplast TMJ disc implants. J Oral Maxillofac Surg 1992;50:133. 4. Bronstein SL. Retained alloplastic temporomandibular joint implants: A retrospective study. Oral Surg 1987;64:135. 5. Chossegros C, Guyot L, Cheynet F, et al. Full-thickness skin graft interposition after temporomandibular joint ankylosis surgery: A study of 31 cases. Int J Oral Maxillofac Surg 1999;28:330. 6. Miyamoto H, Kurita K, Ogi N, et al. The role of the disk in sheep temporomandibular joint ankylosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;88:151.
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7. Lei Z. Auricular cartilage graft interposition after temporomandibular joint ankylosis surgery in children. Oral Maxillofac Surg 2002;60:985-987. 8. Tucker MR, Kennady MC, Jacoway JR. Autologous auricular cartilage implantation following dissectomy in the primate temporomandibular joint. J Oral Maxillofac Surg 1990;48:38.