European Journal of Oral Surgery by Ariesdue srl

Official journal of the SocietĂ Italiana Specializzati in Chirurgia Odontostomatologica ed Orale Vol. 5 issue 2 October 2014 ISSN 2037-7525

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European journal of oral surgery Official journal of the Società Italiana Specializzati in Chirurgia Odontostomatologica ed Orale

www.ejos.eu

Editor-in-chief

Publisher

Prof. Carlo Maiorana (Italy)

Editorial Manager Dr. Federico Mandelli (Italy)

Associate Editors Dr. Filippo Fontana (Italy) Dr. Marco Cicciù (Italy)

Editorial Board Dr. Giovanni Battista Grossi (Italy) Prof. Alan Herford (USA) Prof. Fouad Khoury (Germany) Dr. Paolo Ghensi (Italy) Prof. Aldo Bruno Giannì (Italy) Prof. Jaime A. Gil (Spain) Dr. Mattia Pramstraller (Italy) Prof. Massimo Simion (Italy) Prof. Anton Sculean (Switzerland) Prof. Tiziano Testori (Italy) Prof. Leonardo Trombelli (Italy) Dr. Istvan Urban (Hungary)

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Vol. 5 issue 2 October 2014

page 25

Dental implants maintenance: an observational study on 200 patients

page 31

Dentinogenic ghost cell tumour presenting as an asymptomatic gingival swelling in maxillary anterior region: A case report

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European journal of oral surgery Official journal of the SocietĂ Italiana Specializzati in Chirurgia Odontostomatologica ed Orale

Editorial Dear colleagues,

Prof. Carlo Maiorana Editor-in-chief

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the present issue of eJOS comes together with the third national Congress of Siscoo Society. We are glad that many members showed their appreciation for the program. This congress marks the beginning of a new scientific partnership with the Cenacolo Odontostomatologico Milanese, whose tradition in organizing high level events in the field of Dentistry is very well known in Italy. Thanks to this agreement, every scientific event promoted by Siscoo will be in the future open to the members of the Cenacolo at special conditions and the same behavior will be followed by Cenacolo in respect to Siscoo members. In our eyes, this approach should be very welcome because it means the opportunity to have access to a wider scientific offer. I take this opportunity to thank the President of Cenacolo Milanese, dr Federica Demarosi and the members of the Board, who, with readiness and willingness, accepted my proposal for cooperation. I wish all Siscoo members to enjoy the Congress and to take advantage of this new cooperation between our society and Cenacolo Milanese.

European journal of oral surgery Official journal of the Società Italiana Specializzati in Chirurgia Odontostomatologica ed Orale

Editorial

Dr. Federica Demarosi President Cenacolo Odontostomatologico Milanese

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Dear colleagues, I am pleased to inform you that the meeting on Implantology and Oral Surgery of Cenacolo Odontostomatologico Milanese will be held on February 27th and 28th 2015 in Milan (at the Auditorio San Paolo, via Giotto). This 27th edition of “Giornate Milanesi di Implantologia e Chirurgia Orale” will focus on “minimally invasive versus traditional approach in oral surgery and implantology.” This year the event is organized with the invaluable assistance of Siscoo (Italian Society of Specialists in Dental and Oral Surgery) and its president, prof. Carlo Maiorana. The event is organized in different sections, where two speakers will compare conventional and minimally invasive techniques in the various fields of oral and implant surgery. Prof. Romeo and dr. Leghissa will report on great sinus lift, whereas prof. Francetti and dr. Ricci will compare traditional periodontal treatment with the latest surgical techniques. Then prof. Rasperini and prof. Re will debate on treatment of periodontally compromised teeth versus their replacement with dental implants. As usual, the meeting will devote a section to dental hygienists, this year with Dr. Boldi (president of the Association of Italian dental hygienists) who will describe the use of laser in minimally invasive maintenance of peri-implant tissues, and to dental assistants, with Ms.s Terzo (SIASO vice-president) reporting on risk management in dentistry. The meeting will also include a session where filmed surgical procedures will be commented and discussed by the surgeon, as well as our customay focus session, which this year is devoted to oral oncology and held by prof. Campisi (vicepresident of the Italian Society of Oral Pathology and Medicine). Eventually, let me remind you that the meeting is addressed, as usual, to dentists, dental hygienists, assistants and technicians, in order to emphasize the wish of Cenacolo Odontostomatologico to educate the whole dental team.

EDITORS-IN-CHIEF Adriano Piattelli University of Chieti Pescara (Italy) Arthur Belem Novaes Jr. University of S達o Paulo (Brazil)

w w w.journalofosseointegration.eu

J O U R N A L O F OSSEOINTEGRATION

ISSN 2036-413X GIUGNO 2014 N. 2 VOL. 6 www.journalofosseointegration.eu

cover_02_2014.indd 1

01/07/14 11:40

Printed Italian version

Digital international journal in english language indexed in and

available on

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Monograph

Dental implants maintenance: an observational study on 200 patients Giulia Attardo Giovanni Battista Grossi* Gianluca Bassi** Davide Rancitelli*** Fabrizio Signorino** Carlo Maiorana**** Department of Dental Implants. U. O. C. Maxillofacial Surgery and Odontostomatology, Fondazione IRCCS CĂ Granda. University of Milan, Milan, Italy *MD, DDS **DDS ***DDS, PhD fellow ****MD, DDS. Professor and Chairman, Oral Surgery and Department of Dental Implants

Aim Materials and methods

Results

Conclusion

Aim of this study was to analyze the efficacy of oral hygiene procedures and devices in the prevention of peri-implant disease. The use of oral hygiene maneuvers and their typology were investigated in two hundred patients, with at least one implant placed. Data about frequency and duration of domiciliary dental care were collected together with risk factors and anamnestic data too. Plaque and bleeding indices were used to evaluate the oral status of the sample and, sequentially, a statistical analysis was performed. The results demonstrate that patients who used oral hygiene devices (interdental brush, dental floss) associated to toothbrush show lower plaque index values. The simultaneous use of different devices achieves better results than the use of a single one. No significant differences were noticed between the single use of each device. The use of the oral hygiene devices analyzed and presented in this study, alone or in combination, could be recommended in order to prevent peri-implant disease.

Scatterplot: Mean leeding vs. Mean Plaque Correlation: r = .2206

45% 120

100

35%

Mean Plaque (%)

Plague Index (%)

40%

30%

25% 20

20%

<10 die

>10 die

-20 -20

Tobacco use

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Mean Bleeding (%)

100

120

0,95 Conf. Int.

Key words: Dental Implants, Oral hygiene devices, Bleeding on probing, Plaque Index

Attardo G. et al.

Introduction

Study population and methodology

The placement of osseointegrated implants is becoming one of the most predictable and efficient treatments in the rehabilitation of partially or fully edentulous patients. However, even if in the recent years an increase of both the implant survival and success rates was observed, the pathological conditions that may lead to implant loss should not be underestimated. Implant failure could occur early after surgery, due to unsuccessful osseointegration process, or later for biological or bio-mechanic reasons. Many authors demonstrated that the most frequent cause of implant loss is the onset of peri-implant disease, strictly related to the infection of peri-implant tissues (1, 2). Dental implants, in fact, are characterized by surfaces, often rough, that are potentially colonized by bacteria harbored in the oral cavity. In order to determine the long-term success of the implant therapy, it is mandatory to identify the early signs of disease through the continuous clinical evaluation and maintenance of the patient, assessing the main risk factors and planning correct interventions validated by the most recent evidence-based medical literature. Biological processes leading to implant failure may be slow and gradual. The use of appropriate periodontal parameters and index systems, in order to convert personal impressions into digital data, could help the clinician to control in detail the progression of diseases and copare the therapeutic algorithms stated by the international guidelines. The parameters routinely used during maintenance therapy of patients treated with implants, should be enough sensitive and allow detection of early changes. Many clinical signs of failure, in fact, appear only when an advanced step of disease has already been reached (3). Routinely periimplant evaluation and professional oral hygiene session program seem to be the most important procedures to prevent peri-implant disease. However, it is not possible to achieve good level of oral hygiene only performing professional hygiene. For this reason, patient motivation and home dental hygiene are parameters to focus on, even before planning the surgical step. Toothbrush and dental floss should be used according to oral hygiene instructions given by the clinician; however, considering the advanced average age of edentulous patients and the difficulty to learn or perform appropriate oral hygiene techniques, the use of interdental brush and dental floss is worth further consideration. Aim of this research was to compare the efficacy of oral hygiene procedures and devices, by evaluating bleeding on probing and plaque index on dental implants (4-5).

Selection of the sample In this study, 200 patients were evaluated. All the collected data remained anonymous. All patients were fully informed about the purpose of the study and informed consent was obtained. Data collection was carried out from November 2012 to December 2013.

description of protocol For each patient a medical history form was completed and Plaque index (PI) and Bleeding on Probing (BOP) were recorded in order to compare them with their oral hygiene habits, both at home and professional.

Modified Plaque Index Plaque is considered as an important etiological factor in peri-implantitis development. It is therefore appropriate to assess oral hygiene through a quantitative method. The original PI has been slightly modified to asses plaque formation in the marginal area on ITI implants (mPl) (Table 1) (6). The plaque index was recorded in 4 sites only around implants by circumferential probing with a special plastic probe. The 4 dental sites considered were: buccal, mesiobuccal, distobuccal and lingual/palatal. The 4 measurements taken were summed and divided by 12 (maximum number given by the sum of the siteâ&#x20AC;&#x2122;s values of the highest PI score) in order to obtain the Pl for the single element. The PI for subject was then calculated as the average of the index of the individual implant site. (Mean: sum of the indices of the individual implant sites divided the number of sites considered).

Gingival Bleeding Index This Gingival Bleeding Index, introduced by Ainamo and Bay (1975), is performed through a gentle probing of the orifice of the gingival crevice. If bleeding occurs within 10 seconds, a positive finding is recorded and the number of positive sites is recorded and then expressed as a percentage of the number of sites examined. Bleeding can also function as a motivating factor in spurring patients to improve their oral home care. A total average percent of bleeding index for single patient was given: it takes into account the values of all dental implants (4).

Statistical analysis Statistical analysis was performed using the Statistical Package for Social Sciences (version 21.0, SPSS Inc.,

Score 0

No detection of plaque.

Score 1

Plaque only recognized by running a probe across the smooth marginal surface of the implant.

Score 2

Plaque con be seen by the naked eye.

Score 3

Abundance of soft matter.

tabLE 1 Assessment of plaque accumulation by a modified Plaque Index (mPlI).

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Monograph Indipendent variables

Category

Total number

Plaque Index (%)

Age (years)

<54

33.5

27.2±20.0

average 57

54-61

30.5

25.4±17.1

range 23-83 Gender

Tobacco use

Professional oral hygiene (n° session/year)

Frequency of domiciliary dental care

Duration of domiciliary dental care (min.)

Oral hygiene devices (n°)

Type of oral hygiene devices

>61

36.0

30.5±20.9

42.0

28.9 ±19.6

116

58.0

27.1 ±19.5

140

70.0

24.8±19.4

<10

4.5

33.2±13.2

>10

25.5

35.2±19.1

11.0

31.5±18.2

30.0

25.1±19.7

41.0

27.2±19.8

9.0

34.8±21.7

9.0

28.3±16.6

4.5

45.2±20.4

105

52.5

28.2±20.4

43.0

25.5±17.6

18.5

33.8±21.9

163

81.5

26.5±18.8

40.0

30.8±19.8

45.0

26.5±19.9

30.0

23.9±17.0

None

47.6

30.8±19.8

Interdental Brush

29.2

27.3±18.0

Dental Floss

23.2

24.7±22.4

p-Value 0.31

0.52

0.003

0.36

0.01

0.04

0.17

0.27

tabLE 2 Descriptive statistics and analysis of variance (one- way ANOVA) of independent variables.

Chicago, Illinois, USA). Recorded data were used for calculations of mean values and standard deviations. One way ANOVA test was used to to measure the association between each indipendent variable and the outcome variable (Plaque Index), followed by the post-hoc least significant difference (LSD) test for intergroup differences. The Pearson coefficient was used to measure correlations between the clinical measurements. P values less than 0.05 were considered to be statistically significant.

45%

Plague Index (%)

40%

Results 200 patients (84 females, 116 males) aged 23 to 83 years (average age 57 years) were entered into this study. The descriptive statistics for the sample are summarized in Table 2.

30%

25%

20%

Tobacco use Significant differences in the amount of plaque were noted between the three groups of smokers (p =.003). Post hoc analysis revealed a statistically significant difference between heavy smokers group (>10 cigarettes a day) compared to the no smokers group (p =.001), but no statistically significant differences were observed between the other groups (Fig. 1).

35%

<10 die

>10 die

Tobacco use

fig. 1 Relationship between tobacco use versus PI.

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Attardo G. et al. fig. 2 Scatterplot representing the correlation between mean plaque and mean bleeding (Pearson’s r = 0.22; p = .002).

Scatterplot: Mean leeding vs. Mean Plaque Correlation: r = .2206

120

100

Mean Plaque (%)

-20 -20

Mean Bleeding (%)

100

120

0,95 Conf. Int.

Professional oral hygiene

Type of oral hygiene device

From the sample of 200 patients, 22 patients had not undergone any professional oral hygiene session with a mean of PI around the implants of 31.5% (± 18.2), 60 patients underwent a single yearly session showing a PI of 25.1% (±19.7), 82 patients a two yearly sessions with PI of 27.2% (± 19.8), 18 patients three sessions with PI of 34.8% (± 21.7), 18 patients four sessions with PI of 28.3% (± 16.6). No significant differences in the amount of PI were noted between the five groups.

The comparison between the three groups showed no significant difference, even though dental floss showed the lowest PI values. Finally, a significant correlation was found between PI and BoP (p=.002) (Fig. 2).

Frequency of domiciliary dental care

The most frequent reason of implant failure could be attributed to peri-implant infections. Peri-implant infections are generally classified as peri-implant mucositis and periimplantitis depending on the severity, reversibility and loss of supporting bone (1). The phlogosis of the soft tissues surrounding an implant is evidenced in both the pathologies. Periimplantitis, however, may not develop in all periimplant sites with mucositis, just as periodontitis may not develop in all sites with gingivitis (6). According to Fardal and Grytten, the cost of maintaining implants is much higher than the cost of maintaining teeth (7). Several studies proved a correlation between the bacterial flora observed in periodontal and peri-implant tissues (8, 9). Furthermore, mucositis and periimplantitis were associated to the same bacteria that induce respectively gingivitis and periodontal disease, while the flora associated with healthy peri-implant tissue seems to be similar to the microbiota related with healthy gingiva (9). An adequate plaque control performed by the patient is basic to avoid the onset of infections both in teeth and in dental implants (10, 11). In long term studies, patients with good oral hygiene tended to keep implants longer. With adequate oral hygiene practices, the presence of keratinized peri-implant mucosa appears not to be essential for the maintenance of

As it is inferred from data, a reduction of 17% of PI between patients that perform domiciliary dental care once and twice daily has been found. This difference was statistically significant (p=.01).

Duration of domiciliary dental care The analyzed sample was composed of 37 patients who performed sessions of domiciliary dental care in less than one minute and of 163 patients declaring to dedicate more than one minute to domiciliary dental care. The percentage of PI in the first group resulted 33.8% (±21.9%), while the second group showed PI of 26.5% (± 18.8%). This difference was statistically significant (p=.04).

Number of oral hygiene devices 80 patients did not use any oral hygiene devices presenting a PI of 30.8% (± 19.8), 90 used a single oral hygiene device presenting a PI value of 26.5% (± 19.9) and 30 patients used two oral hygiene devices showing a PI of 23.9% (± 17.0). The differences between the three groups were not statistically significant.

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Discussion

Monograph implant stability (1). The only successful option to prevent plaque formation is represented by the mechanical removing performed by different oral hygiene devices. Many authors investigated the correct techniques and maintenance protocol for dental implants (12-15). Patients should be evaluated at regular intervals to monitor their peri-implant status, the condition of implant-supported prostheses, and plaque control. The key role of home oral hygiene is universally known. The evaluation of oral hygiene and patient motivation prior to the implant placement is indicated; imparting clear oral hygiene instructions and a good motivation is paramount (16). Clinicians should pay attention to communicate to the patient the importance of home care, and be sure to be completely understood. Home care instructions should be customized according to implant design and accessibility (17). Several oral hygiene devices were designed and produced for different dental areas and different patients. The interdental space is one of the most difficult areas to be cleaned, especially for elderly people. There are several devices designed to operate in an easy and efficient way: dental floss, single and interdental brushes. Two of the most useful and indicative parameters to investigate peri-implant disease are BoP and PI. The present study focused attention on the evaluation of these two indices specifically on dental implants. In the present study a strict correlation between the presence of plaque and bleeding was found; in fact, plaque represents a reservoir of bacterial pathogens for focal infections, which activate a local inflammatory response. Nevertheless, Newcomb et al. demonstrated that it is not possible to relate a specific bacterial assay to clinical signs such as bleeding on probing or suppuration (18). The present study also investigated whether there was correlation between PI compared to the consumption of cigarettes. A statistically significant difference was detected only in the plaque index of heavy smokers. In a previous study performed by Bastiaan and Waite plaque levels appeared to be higher in smokers than nonsmokers, even though the differences were not statistically significant (19). No consistent differences were evident in the gingival status of the two groups, according to the present study. Smokers group showed a higher percentage of Gram + bacteria than nonsmokers in the first three days of evaluation. Muller et al. according to our results, examining 65 patients, recorded an increase of supragingival plaque and calculus in heavy smokers group, both at time 0 and 6 months, while comparing the single sites, lower values of BoP were observed (20). This figure is probably related to the reduction of vascularization due to the action of smoking. At the beginning of implantology, the key for long-term success of osseointegrated implants was the surgical phase. In more recent years, clinicians recognized professional implant maintenance and diligent patient home care as two critical factors for the long-term success of dental implants (15). The adoption of a systematic hygienic protocol is effective in keeping low the incidence of peri-implant mucositis as well as in controlling plaque accumulation and clinical attachment loss (10). As far as plaque and bleeding are concerned, this study inferred that professional hygiene does not significantly affect in terms of frequency and duration if compared to the importance of the accuracy of domiciliary dental care. Furthermore, even the study of Kracher et al. suggested that the negation of early microbial accumulation on the

dental implant surfaces and the elimination of at least 85% of plaque biofilm by the patient are crucial for a long-term success (21). Another point addressed was to identify which of the oral hygiene aids to be associated with brushing, estimated on the strength of reduction of plaque and bleeding, would allow a better compliance of the patient with implantprosthetic rehabilitation. In the studies found in the literature on natural teeth, it has been found that the use of dental floss associated with brushing would permits a reduction of bleeding of 50% aside from type of floss used (waxed, flat, spongy) (22]). Also Ong et al., in a comparative study, did not observe statistically significative differences between three different kinds of dental floss (12). Over the years, it has been generally accepted that dental floss has a positive effect on removing plaque and decreasing bleeding (911). The American Dental Association (ADA) even reports that up to 80% of plaque may be removed by this method. Several reviews have been conducted on the effectiveness of different procedures and devices dedicated to interdental space, however, only few reviews are systematic and none of them has conducted a meta-analysis. Also, a limited number of papers provided data on the efficacy of flossing and tooth brushing compared to tooth brushing alone. Warren et al. demonstrated that flossing in association with tooth brushing produced no clear benefits (23). Additionally, a recent review showed that self-flossing has no effect on reducing caries risk (5). The present study did not evidence statistically significant differences between dental floss and interdental brush on dental implant, even though dental floss seemed to show lower PI values. Interdental brushes commercially available have different shapes and designs. Jordan et al. in a randomized controlled trial concluded that straight interdental brushes might better remove plaque interproximally when compared to angled interdental brushes (24). The recent literature highlighted also how the use of interdental brush seems to guarantee better performance expressed as lower PI and BoP values on natural teeth (25-27). De Slot et al., in a systematic review, analyzed the effects of the use of interdental brush associated to toothbrush (25). Not only they observed that interdental brush removes more dental plaque than brushing alone, but also that in most studies had a positive significant difference on PI when compared with dental floss. Christou et al. affirmed that the use of interdental brushes is more effective in plaque removal and results especially in a larger reduction of probing depth than the use of dental floss (27). Even observing small differences between the groups, according to patient preferences, they concluded that interdental brushes should be considered preferable to floss. The data of this study do not prove that an aid of oral hygiene was more effective than another; also the study of 2010 of Kracher et al. emphasized how domiciliary dental care is dictated by the prosthetic design (location and angulation of the implants, the length and the position of the transmucosal abutments) and that the choice of the most suitable device depends on the indications of each single patients (21).

Conclusion According to the results of this study, it may be concluded

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Attardo G. et al. that interdental brush and dental floss, associated to toothbrush, remove more dental plaque than brushing alone. No significant differences were observed in the comparison between the various devices. The communication with the patient is still the most important valuable step: analyzing risk factors and underlining the importance of motivation. The clinician should choose on the basis of the prosthetic design and patient compliance.

References 1. Lang NP, Wilson TG, Corbet EF. Biological complications with dental implants: their prevention, diagnosis and treatment. Clin Oral Implants Res 2000;11 Suppl 1:146-55.첫 2. Salvi GE, Lang NP. Diagnostic parameters for monitoring peri-implant conditions. Int J Oral Maxillofac Implants 2004;19 Suppl:116-27. 3. Mombelli A, Lang NP. Clinical parameters for the evaluation of dental implants. Periodontol 2000 1994;4:81-6 4. Silverstein L, Garg A, Callan D, Shatz P. The key to success: maintaining the long-term health of implants. Dent Today 1998;17:104 5. Position paper: tobacco use and the periodontal patient. Research, Science and Therapy Committee of the American Academy of Periodontology. J Periodontol 1999;70:1419-27. 6. Ainamo J, Bay I. Problems and proposals for recording gingivitis and plaque. Int Dent J 1975;25:229-35. 7. Fardal O, Grytten J. A comparison of teeth and implants during maintenance therapy in terms of the number of disease-free years and costs - an in vivo internal control study. J Clin Periodontol 2013;40:645-51. 8. Pontoriero R, Tonelli MP, Carnevale G, Mombelli A, Nyman SR, Lang NP. Experimentally induced peri-implant mucositis. A clinical study in humans. Clin Oral Implants Res 1994;5:254-9. 9. Lang NP, Bragger U, Walther D, Beamer B, Kornman KS. Ligature-induced peri-implant infection in cynomolgus monkeys. I. Clinical and radiographic findings. Clin Oral Implants Res 1993;4:2-11. 10. Serino G, Strom C. Peri-implantitis in partially edentulous patients: association with inadequate plaque control. Clin Oral Implants Res 2009;20:169-74.

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11. Corbella S, Del Fabbro M, Taschieri S, De Siena F, Francetti L. Clinical evaluation of an implant maintenance protocol for the prevention of peri-implant diseases in patients treated with immediately loaded full-arch rehabilitations. Int J Dent Hyg 2011;9:216-22. 12. Jensen RL, Jensen JH. Peri-implant maintenance. Northwest Dent 1991;70:14-23. 13. Lord BJ. Maintenance procedures for the implant patient. Aust Prosthodont J 1995;9:33-8. 14. Chen S, Darby I. Dental implants: maintenance, care and treatment of peri-implant infection. Aust Dent J 2003;48:212-20. 15. Abubakar A, Van Baar A, Fischer R, Bomu G, Gona JK, Newton CR. Socio-cultural determinants of health-seeking behaviour on the Kenyan coast: a qualitative study. PLoS One 2013;8:e71998. 16. Bader HI. How to motivate, inform dental implant patients on home care. Dent Implantol Update 1993;4:57-60. 17. Todescan S, Lavigne S, Kelekis-Cholakis A. Guidance for the maintenance care of dental implants: clinical review. J Can Dent Assoc 2012;78:c107. 18. Newcomb GM, Nixon KC. The relationship between microbiological assays and the clinical signs of periodontal disease. Aust Dent J 1989;34:13-9. 19. Bastiaan RJ, Waite IM. Effects of tobacco smoking on plaque development and gingivitis. J Periodontol 1978;49:480-2. 20. Muller HP, Stadermann S, Heinecke A. Longitudinal association between plaque and gingival bleeding in smokers and non-smokers. J Clin Periodontol 2002;29:287-94. 21. Kracher CM, Smith WS. Oral health maintenance dental implants. Dent Assist 2010;79:27-35. 22. Graves RC, Disney JA, Stamm JW. Comparative effectiveness of flossing and brushing in reducing interproximal bleeding. J Periodontol 1989;60:243-7. 23. Warren PR, Chater BV. An overview of established interdental cleaning methods. J Clin Dent 1996;7:65-9. 24. Jordan RA, Hong HM, Lucaciu A, Zimmer S. Efficacy of straight versus angled interdental brushes on interproximal tooth cleaning: a randomized controlled trial. Int J Dent Hyg 2014;12:152-7. 25. Slot DE, Dorfer CE, Van der Weijden GA. The efficacy of interdental brushes on plaque and parameters of periodontal inflammation: a systematic review. Int J Dent Hyg 2008;6:25364. 26. Kiger RD, Nylund K, Feller RP. A comparison of proximal plaque removal using floss and interdental brushes. J Clin Periodontol 1991;18:681-4. 27. Christou V, Timmerman MF, Van der Velden U, Van der Weijden FA. Comparison of different approaches of interdental oral hygiene: interdental brushes versus dental floss. J Periodontol 1998;69:759-64.

Case report

Dentinogenic ghost cell tumour presenting as an asymptomatic gingival swelling in maxillary anterior region: A case report Manuel Suvy* L.K. Surej Kumar** J. Nair Bindu*** ***Menon Varun P.*** Department of Oral and Maxillofacial Surgery - PMS College of Dental Science, Trivandrum, India *BDS, MDS, DNB, MOS RCS(Ed), MFDS RCS (Eng) Oral and Maxillofacial Surgeon, Associate Professor **BDS, MDS, Oral and Maxillofacial Surgeon, Professor and Head ***BDS, MDS, Oral and Maxillofacial Pathologist Professor and Head Dept of Oral and Maxillofacial pathology PMS College of Dental Science, Trivandrum, India ****Junior resident Oral and Maxillofacial Surgeon

Background

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The Dentinogenic ghost cell tumor comes under the spectrum of ghost cell lesions. Uniqueness and rarity of the lesion is evident from the literature. Here we are presenting a case report of dentinogenic ghost cell tumor which clinically presented as an asymptomatic gingival swelling in the maxillary anterior region along with its diagnosis, management and review of literature.

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Key words: Dentinogenic ghost cell tumor, Maxilla, DGCT, Ghost cell

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Introduction

fig. 1 Clinical photograph showing swelling in the right anterior maxillary region.

Dentinogenic ghost cell tumor is a locally invasive rare neoplasm, seen in any age group from 10 to 90 years, and there is no significant difference between genders. Dentinogenic ghost cell tumor occurring in the mandible and maxilla is called central or intraosseous, whereas in the alveolar mucosa and gingival soft tissues is called peripheral or extraosseous. The great majority of these tumours are benign and are treated by local resection. The uniqueness of the lesion is quite evident from the literature. Here we report a case of a dentinogenic ghost cell tumor clinically reported as an asymptomatic gingival swelling in the maxillary anterior region with its diagnosis and treatment.

fig. 2 OPG showing radiolucent lesion involving upper right lateral incisor and canine region.

Case report Case history A 40 year old female underwent clinical examination at the oral and maxillofacial surgery department with complaints of multiple decayed teeth. Clinical examination revealed an incidental finding of gingival swelling approximately of 2 x 3 cm in size, in relation to 12, 13 region. It was noticed that the swelling was oval shaped, diffuse, extending from the mesial aspect of 11 to the distal aspect of 14, and superiorly it extended into the sulcus (Fig. 1). Overlying gingiva showed a localized gingival fibromatosis. Case history revealed that the patient had noticed the swelling 78 years before, and that there were no symptoms associated with its growth. Intraoral periapical radiograph showed a well defined circumscribed lesion, of size 1 x 1 cm with sclerotic borders in the region of 12, 13. Vitality test revealed that the teeth involved were vital. Orthopantomograph showed well defined oval shaped radiolucency in relation 12, 13 region (Fig. 2).

Differential diagnosis The patient presented with a history of swelling noticed for the previous 7-8 years in the anterior region of the mandible with no associated symptoms. Following lesions were considered in the differential diagnosis of this slow

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growing, asymptomatic swelling on the basis of its position, clinical features and radiological features: Peripheral ossifying fibroma, Globulomaxillary cyst, Lateral periodontal cyst, Adenomatoid odontogenic tumour, Squamous odontogenic tumour, Peripheral odontogenic fibroma and Peripheral ameloblastoma. Peripheral ossifying fibroma is a reactive lesion that only occurs on the gingiva, most commonly in the maxillary anterior region. The peak incidence is between the second and third decades. Women are more likely to be affected than men. Lesions range in color from pink to red, and the surface may or may not be ulcerated. It is seen as asymptomatic well-circumscribed, pale pink, nodular growth which is non tender and firm to hard in consistency. It has a slight predilection for the maxillary gingiva (1). The radiographic features may range from no changes to, in rare instances, superficial bone erosions. Globulomaxillary cysts classically develop between maxillary lateral incisor and cuspid teeth, although occasionally reported between central and lateral incisors. Radiographs typically demonstrate a wellcircumscribed unilocular radiolucency between the teeth. This lesion has a distinct appearance which is an oval, round, or inverted pear-shaped

radiolucency between the roots of the lateral incisor and cuspid teeth, causing divergence (1). It is usually seen in young adults. The radiological picture was not similar to that of a lateral periodontal cyst and also they usually present as soft tissue swellings below the interdental papillae. For these reasons lateral periodontal cyst was ruled out from the differential diagnosis. Adenamatoid Odontogenic tumor is largely limited to younger patients, it has a striking tendency to occur in anterior portions of the jaws and is found twice in the maxilla than in the mandible .Females are affected twice as often as males. Most of these lesions are relatively small. The peripheral forms of the tumor are small sessile masses on the facial gingiva of the maxilla. Clinically these lesions cannot be differentiated from the common gingival fibrous lesions (1). Squamous odontogenic tumor, found in ages ranging from 8 to 74 years, is randomly distributed throughout the alveolar process of maxilla and mandible with no site or sex predilection. A painless or mildly painful gingival swelling is often associated with the mobility of the involved teeth. About 25% of reported patients have had no symptoms. The radiographic findings are not specific or diagnostic (1).

Case report fig. 3 Intraoperative photograph showing the cystic nature of the lesion once the overlying cortex has been removed.

Although peripheral odontogenic tumors are rare when compared with their intraosseous counterparts, a differential diagnosis of a localized gingival mass would be incomplete without their inclusion. Several varieties of these tumors have been reported in the literature, the most common ones being the peripheral odontogenic fibroma and the peripheral ameloblastoma, usually seen in age groups between 23-82 years. The condition usually presents as slow-growing, pink to red, sessile or pedunculated, firm, well-defined, asymptomatic nodules of the attached gingiva. An accurate diagnosis of these lesions is solely dependent on microscopic examination of the excised gingival mass as radiologic features are not seen in most cases (1, 2).

fig. 4 Surgical bed demonstrating the defect and bone loss at the mesial region of canine root, maxillary region.

Surgical management A two sided full thickness trapezoidal mucoperiosteal flap was elevated, extending within the area from tooth 15 to 21. The lesion was identified in relation to 14, 13, and 12. Clinically it was seen that the labial cortical plate was perforated at some levels. The cystic lining was clearly evident (Fig. 3), once the overlying bone was removed .Following the enucleation it was seen that tooth13 had mild root resorption on the mesial aspect (Fig. 4). The excised specimen was sent for histopathologic examination.

moderately collagenous connective tissue with odontogenic epithelium, clusters of ghost cells undergoing

dystrophic calcification and eosinophilic areas suggestive of dentinoid (Fig. 5), the latter were seen in close contact

Diagnosis The

soft

tissue

section

showed

fig. 5 Moderately dense collagenous stroma showing large areas of dentinoid and interconnecting strands of odontogenic epithelium. (H & E X100).

fig. 6 Areas of dentinoid with ghost cells undergoing calcification and odontogenic epithelium (H & E X 400).

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fig. 7 Clusters of ghost cells showing calcification seen in association with odontogenic epithelium and dentinoid (H & E x 400).

fig. 8 Proliferating odontogenic epithelium (H & E X 400).

fig. 9 Van Gieson stain showing (x 400). a) ghost cells staining yellow. b) Surrounding dentinoid staining pink. c) odontogenic epithelium.

with the odontogenic epithelium and ghost cells (Fig. 6, 7). The odontogenic epithelium was arranged in interconnecting cords and islands and was composed of low columnar and spindle cells (Fig. 8). In some portions of the tumour, irregular areas of calcified material were seen. Adipose cells in the connective tissue and mature bone in the periphery were also observed. The histopathological picture gave an impression of Dentinogenic ghost cell tumor. It was decided to study more sections of the excised specimen using special stains to confirm the diagnosis. Van Gieson staining was done and the ghost cells took a yellow stain and dentinoid pink (Fig. 9). The asymptomatic swelling was confirmed as Dentinogenic ghost cell tumor (DGCT).

Discussion The rarity of the occurrence of DGCT as well as the cystic appearance of the lesion mislead us, thus not including it in our differential diagnosis. As for the classification of ghost cell odontogenic tumours (3), only CCOT type 1 is a cystic form and not the dentinogenic

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Case report ghost cell tumour, so the biopsy report came quite as a surprise. Hence this appears to be a unique case of DGCT which presented in cystic form. DGCT currently comes under the spectrum of ghost cell lesions, and Calcifying Odontogenic Cyst (COC) is one of the earliest described ghost cell lesion: it was first described by Gorlin et al. in 1962 (4). It represents about 1% of all odontogenic cysts. COC tends to occur around the third decade of life, though patients age ranges from 7 to 82 years (5, 6). It occurs equally in the maxilla and mandible, usually anterior to the first permanent molar, though it has a predilection for the maxilla in the younger age range. It occurs equally in males and females. It exists in two entities: cystic tumor or solid neoplasm (5, 6) In 1981, Praetorius et al. proposed a classification which resolved the dilemma between the cystic or neoplastic nature of calcifying odontogenic cyst. He recognized four different histological patterns of COC and classified them as type 1A (simple unicystic), type 1B (odontome-producing), type 1C (ameloblastomatous proliferating) and type 2 (dentinogenic ghost cell tumour) (5). According to a recent WHO classification of odontogenic tumours, COC was re-named as calcifying cystic odontogenic tumour (CCOT). Ghost cell odontogenic tumours were classified into three entities (7). The first entity CCOT is characterized by a well-defined, cystic, painless, slowly growing neoplasm that rarely relapses. The second, the DGCT, is a locally invasive neoplasm which shows more aggressive behavior and recurrence. The third entity is a malignant counterpart of CCOT and DGCT, named as ghost cell odontogenic carcinoma (GCOC) (3). Dentinogenic ghost cell tumor (DGCT) is an extremely rare odontogenic tumor. According to Buchner’s (8) research on 215 cases of intraosseous calcifying odontogenic cysts, calcifying odontogenic cysts (COCs) account for 1-2% of all odontogenic tumors and only 2-14% of COCs are solid tumors, the latter were considered to be DGCTs. Thus, based on even a large sample size of patients that may have DGCTs, it remains a rare finding. Dentinogenic ghost cell tumor (DGCT) was first described by Fejerskov and Krogh (9) in 1972. They had used the

term “calcifying ghost cell odontogenic tumor”; but in 1981, Praetorius et al. (5) suggested the term “dentinogenic ghost cell tumor”. Shear (10) in 1983 gave the term “dentinoameloblastoma” because of its similarities with ameloblastoma and dentinoid production. In 1986, Ellis and Shmookler (6) proposed the term “epithelial odontogenic ghost cell tumor” as they thought that the ghost nucleated keratinizing cell was the most distinctive histopathological feature. It was Hong et al. (11) in 1991 who suggested the term “epithelial odontogenic ghost cell tumor” as characteristic of these neoplasms are the odontogenic epithelial proliferations with some inductive activity and the formation of ghost cells. In 2003, Li and Yu (12) suggested the term “odontogenic ghost cell tumor” which was originally described by Ellis (13) in 1999. This term emphasized its origin, neoplastic nature, and most striking histopathological features. In 2005, the World Health Organization (WHO) (7) retained the term dentinogenic ghost cell tumor as initially proposed by Praetorius et al. (5). The WHO defined this tumor as “A locally invasive neoplasm characterized by ameloblastoma-like islands of epithelial cells in a mature connective tissue stroma. Aberrant keratinization may be found in the form of ghost cells in association with varying amounts of dysplastic dentin” (7). DGCTs have occurred in patients ranging from 10-92 years of age, with an average age of occurrence of 59 years (14). Two variant forms of DGCT are known to occur; DGCT type1 Central (in the mandible or maxilla) as seen in our case and DGCT type 2 Peripheral (in the gingival; soft tissues or alveolar mucosa). The peripheral variant is the less aggressive of the two types (3). The presence of ghost cells within the proliferative odontogenic epithelium is the essential characteristic for the diagnosis of ghost cell tumours. However, the ghost cells alone are not sufficient for the diagnosis of DGCT nor is it pathognomonic. Ghost cells may also be observed in other lesions such as odontomas, ameloblastomas, and ameloblastic fibro-odontomas (15). The latter tumors were eliminated from the histopathological differential diagnosis by the absence of a cellular primitive ectomesenchyme resembling

dental papilla ,and the presence of dentinoid confirmed the lesion as dentinogenic ghost cell tumour. The treatment involves a surgical excision with a long-term follow up and the recurrence depends on completeness of cyst removal (16). Howerver, in our literature search we came across a case of DGCT in the left body of the mandible, which underwent segmental resection and was reconstructed using an absorbable collagen sponge, rhBMP-2 and a titanium plate and mesh (17). The prognosis of DGCT is determined by the treatment provided. Numerous cases in the literature have been reported to recur, mainly owing to inadequate excision. The retreatment of such cases was found to be totally curative. A review by Kasahara et al demonstrated the rate of recurrence of central DGCTs following resection to be approximately 44% (18). Wide local resection is the recommended mode of treatment for DGCT (19). Till 2009 approximately 32 cases of DGCT have been reported in English literature (19). The malignant transformation of the DGCT after several recurrences has been documented (20). The present case is under routine follow up for the past two years and there is no evidence of any recurrence.

Conclusion Ghost cell tumours are relatively rare and DGCT, being a part of the spectrum is even rarer, and this is the reason for not including it in the initial differential diagnosis. The uniqueness of DGCT is evident in the discussion, as it has been known by a multitude of definitions and multiple classifications. Thus we have presented a case of DGCT with cystic features presenting as an asymptomatic gingival swelling in the anterior maxillary region.

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