CLINICAL DENTISTRY AND RESEARCH 2015; 39(1): 42-47 Case Report

CorrespondenceNaile Cura, DDS, PhD

İzmir CTG Dental Care, Kahramanlar ,İzmir

Telephone: +90 5546325616

E-mail: nailecura@gmail.com

Naile Cura DDS, PhDPrivate Practice

İzmir, Turkey

Serkan Dadakoğlu DDS, PhDPrivate Practice

İzmir, Turkey

Timur Songür, DDS, PhDDepartment of Oral and Maxillofacial Surgery,

Faculty of Dentistry, Turgut Özal University,

Ankara, Turkey

BoTryoiD oDoNTogeNiC CyST: rePorT of A CASe

ABSTrACT

Botyroid odontogenic cyst (BOC) is considered a rare multilocular

variant of the lateral periodontal cyst, usually involves the mandibular

premolar-canine area, followed by the anterior region of the maxilla.

Adults older than 50 years are the most affected group. A 57 year-

old male patient referred to our clinic, Ankara University Faculty

of Dentistry Department of Oral and Maxillofacial Surgery for

evaluation of a swelling in the right anterior mandible. The diagnosis

of a BOC was made based on location and the histopathological

findings of multiple cystic spaces lined by nonkeratinized stratified

squamous epithelium. The 9-month follow-up revealed a normal

clinical appearance with evidence of radiographic bone fill at the site

of the lesion. BOC is known to be a recurrent odontogenic cyst. The

recurrence rate may range between 15% and 20%. The prevailing

opinion is that main reason for recurrence was failure to remove the

entire multilocular lesion during surgery. An extended post-surgical

follow-up is necessary for a patient who has been diagnosed with

BOC.

Keywords: Botryoid Odontogenic Cyst, Lateral Periodontal

Cyst, Radiolucency.

Submitted for Publication: 12.10.2013

Accepted for Publication : 01.22.2015

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CLINICAL DENTISTRY AND RESEARCH 2015; 39(1): 42-47 Olgu Bildirimi

Sorumlu yazarNaile Cura,

Özel İzmir CTG Ağız ve Diş Sağlığı Merkezi ,1416.Sok.

No:34 35230 Kahramanlar, İzmir

Phone: +90 5546325616

E-mail: nailecura@gmail.com

Naile CuraDr., Ağız, Diş ve Çene Cerrahisi Uzmanı

Serbest Diş Hekimi

İzmir, Turkiye

Serkan DadakoğluDr., Ağız,Diş ve Çene Cerrahisi Uzmanı

Serbest Diş Hekimi

Ankara, Türkiye

Timur SongürDr., Turgut Özal Üniversitesi Diş Hekimliği Fakültesi

Ağız, Diş ve Çene Cerrahisi Anabilim Dalı

Ankara, Türkiye

BOTRYOİD ODONTOJENİK KİST: VAKA RAPORU

ÖZBotyroid odontojenik kist lateral periodontal kistin nadir görülen

multilokuler bir varyantıdır, genellikle mandibular premolar-kanin

dişler bölgesinde ve maksilla anterior bölgede görülür. Lezyon

genellikle 50 yaş sonrası erişkinlerde görülür. Bu olgu sunumunda

57 yaşında erkek hasta, sağ anterior mandibular bölgede şişlik

şikayeti ile Ankara Üniversitesi Diş Hekimliği Fakültesi Ağız,

Diş ve Çene Cerrahisi Anabilim Dalı’na başvurmuştur. Botryoid

odontojenik kist tanısı lokalizasyona ve nonkeratinize çok

katlı yassı epitelle çevrilmiş multiple kistik alanlar şeklindeki

histopatolojik bulgulara dayanarak konulmuştur. 9 aylık post

operatif takip sonrası kist çıkarılan bölgede radyolojik incelemede

gözlenen kemik oluşumu ile birlikte normal klinik görünüm

izlenmiştir. Botryoid odontojenik kist rekürrens oranı %15 ile

%20 arasında değişen nüks ihtimali yüksek bir odontojenik kisttir.

Rekürrensin ana sebebi multilokuler lezyonun geniş cerrahi

eksizyonunun yapılmaması olarak düşünülmektedir. Botryoid

odontojenik kist tanısı konulan hastalarda uzun sureli post

operatif takip önerilmektedir.

Anahtar Kelimeler: Botryoid Odontojenik Kist, Lateral

Periodontal Kist, Radyolusensi

Yayın Başvuru Tarihi : 10.12.2013

Yayına Kabul Tarihi : 22.01.2015

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CLINICAL DENTISTRY AND RESEARCH

INTRODUCTION

A multilocular variant of the lateral periodontal cyst (LPC)

known as the botryoid odontogenic cyst (BOC) has been

identified in 1973 by Weathers and Waldron.1 It has been

suggested the term botryoid odontogenic cyst as an

appropriate descriptive name because of the grapelike

configuration of the gross appearance of these cysts and

their odontogenic origin.1

The most current theory of origin suggests that LPCs and

BOCs arise from the dental lamina, in part because of the

presence of glycogen-rich clear cells in the cyst wall that are

similar to the cells found in dental lamina rests.2

Histologically, both lesions appear the same. The cystic

lining is composed of one to five cell layers of squamous

or cuboidal epithelium with a fibrous connective tissue

according to inflammation.3,4 The epithelium shows areas

of focal thickening with glycogen containing clear cells

observed around the cyst lining.5-8 The BOC and LPC often

have a subepithelial zone of hyalinization.6,9

The odontogenic epithelium responsible for the cystic lining

in the BOC and LPC is hypothesized to arise from three

possible sources: (1) a reduced enamel epithelium (REE), (2)

epithelial cell rests of Malassez (ERMs), or (3) residuals of

dental lamina.4

CASe rePorT

A 57 year-old male patient referred to our clinic, Ankara

University Faculty of Dentistry Department of Oral and

Maxillofacial Surgery for evaluation of a swelling seen in

the right anterior mandible. His systemic diseases include:

diabetes mellitus type 2 and hypertension. He had been

aware of the asymptomatic swelling during the past 8

months. At the time of dental examination, an expansion

behind the right mandibular canine was observed, panoramic

radiography and CT revealed a well-defined unilocular

radiolucent lesion distal to the right mandibular canine in

the edentulous region (Figures 1, 2 and 3). The canine tooth

was devital. There was no paresthesia, tenderness or other

changes in sensation. An excisional biopsy was performed

that included the canine tooth under local anesthesia

(Figures 4, 5 and 6).

The pathology report was indicated as a diagnosis of BOC.

Multiple cystic spaces lined by nonkeratinized stratified

squamous epithelium were seen histopathologically (Figure

7). The cyst lining was thickest layer of the three cell layers

with areas of plaque-like thickening in the epithelium.

Figure1. Bucco-lingual width of the lesion

Figure 2. CT shows unilocular cystic lesion about 15 mm in diameter in the right mandibular region.

Figure 3. Operative view of the intra-oral approach.

The cyst wall was composed of fibrous tissue exhibiting extravasated blood cells without inflammation. At the 9-month follow-up, the clinical appearance of the surgical site was normal (Figures 8, 9 and 10). The patient is using total prosthesis and there is no complaining about the

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RADIOLUCENCY IN MANDIBULA

The enucleation of a BOC or LPC results in an osseous cavity or defect. Lehrhaupt et al.4 treated a residual LPC defect that perforated the facial and lingual mandibular cortex in a similar characterization. A demineralized freeze-dried bone allograft had been used and a membrane had not been placed over the bone graft material. Nart et al.5 reported

Figure 4. Enucleation and tooth extraction

Figure 5. Macroscopic appearance of the surgical specimen.

Figure 6. Post-operative intra-oral photograph.

Figure 7. In the right side of the picture stratified squamoid plaques were shown.

right mandibular region.

DISCUSSION

BOC usually involves the mandibular premolar-canine area, followed by the anterior region of the maxilla.10 The most affected group is adults older than 50 years.11 Although some of the reported cases of BOC have shown a multilocular radiolucency, this image is not characteristic for BOC. There are similarities between BOC and some odontogenic tumors such as ameloblastoma, odontogenic myxoma in terms of appearance. Preoperative differential diagnosis can be carried out by means of incisional biopsy. Some authors demonstrated that this lesion frequently presents as a unilocular radiographic image, thus resembling a variety of other odontogenic cysts or neoplastic conditions.12

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CLINICAL DENTISTRY AND RESEARCH

a similar treatment of a residual LPC osseous defect using guided tissue regeneration with a demineralized freeze-dried bone allograft and resorbable collagen membrane. At the 7-month follow-up, they found significant radiographic bone fill, which was also seen on surgical reentry. Because of the recurrence risk of BOC, wide excision is required for treatment. In the current case, we treated the patient by enucleation.

There is no comment about the state of vital and nonvital adjacent teeth to the lesion in the literature. When a radiolucent lesion is seen related with teeth that is identified on a radiographic examination , histologic examination must be performed if it has a diagnosis about normal, vital pulps.BOC is known as a recurrent odontogenic cyst. The recurrence rate may range between 15% and 20%.13 The histopathological appearance of BOC is quite typical in the presence of epithelial proliferations (plaques). The proliferation rate of the epithelial lining can be determined to estimate the possibility of recurrence. The recurrence risk of BOC is similar to odontogenic keratocyst.The characteristic that distinguishes BOC from LPC is the larger size of the BOC because LPC has a limited growth potential.14 The importance of the differentiation between LPC and BOC is due to the histologic multilocular aspect of the BOC that induces this lesion more expansive. Thereby, increasing the possibility of recurrence because its complete surgical removal is more difficult.9,14

BOC shows some histopathological similarities to the glandular odontogenic cyst (GOC) or sialo-odontogenic cyst (SOC) depending on the presence of mucous cells and surface columnar cells.10,15,16 However, the best classification for these lesions is BOC. These lesions can be seen at opposite ends of a spectrum. The presence of mucous cells does not suspend the BOC from an odontogenic origin, this characteristic of BOC has been reported in a variety of odontogenic cysts such as the dentigerous cyst with a metaplastic phenomenon.17 Furthermore, immunohistochemical studies18,19 have suggested that the GOC is a histological variant of BOC. According to a prevalent opinion, main reason for recurrence can be a failure during the complete removal of the multilocular lesion during surgery. A long term post-surgical follow-up is necessary for a patient whit a diagnosis of BOC.3,13 Alternative treatment techniques for BOC can be considered such as odontogenic keratocyst.11

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2. Hethcox JM, Kirkpatrick TC. Case report: Diagnosis and treatment of a botryoid odontogenic cyst found in the maxillary anterior region. J Endod 2010; 36: 751-754.

3. Altini M, Shear M. The lateral periodontal cyst: an update. J Oral Pathol Med 1992; 21: 245-250.

Figure 8. Post-operative 1-month panoramic radiography

Figure 9. Post-operative 9-month panoramic radiography

Figure 10. Post-operative 9-month intra-oral photograph

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RADIOLUCENCY IN MANDIBULA

4. Lehrhaupt N, Brownstein C, Deasy M. Osseous repair of a lateral periodontal cyst. J Periodontol 1997; 68: 608-611.

5. Nart J, Gagari E, Kahn M et al. Use of guided tissue regeneration in the treatment of a lateral periodontal cyst with a 7-month reentry. J Periodontol 2007; 78: 1360-1364.

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7. Lynch D, Madden C. The botryoid odontogenic cyst. Report of a case and review of the literature. J Periodontol 1985; 56: 163-167.

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10. Padayache A, Vanwyk CW. Two cystic lesions with features of both the botryoid odontogenic cyst and the central mucoepidermoid tumor: sialo-odontogenic cyst? J Oral Pathol 1987; 16: 499-504.

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14. Gardner DG, Kessler HP, Morency R, Schaffner DL. The glandular odontogenic cyst: an apparent entity. J Oral Pathol 1988; 17: 359-366.

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16. Farina VH, Brando AAH, Almeida JD, Cabral LAG. Clinical and histologic features of botryoid odontogenic cyst: a case report. J Med Case Rep 2010; 4: 260.

17. Sciubba JJ, Fantasia JE, Kahn LB. Odontogenic cysts. In: Rosai J, Sobin LH, editors. Atlas of tumor pathology, tumors and cysts of the jaw. Washington DC: AFIP; 2001. p. 15-49.

18. Semba I, Kitano M, Mimura T, Sonoda S, Miyawaki A. Glandular odontogenic cyst: analysis of cytokeratin expression and clinicopathological features. J Oral Pathol Med 1994; 23: 377-82.

19. Koppang HS, Johannessen S, Haugen LK, Haanaes HR, Solheim T, Donath K. Glandular odontogenic cyst (sialo-odontogenic cyst): report of two cases and literature review of 45 previously reported cases. J Oral Pathol Med 1998; 27: 455-462.