Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 3134

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Asian Journal of Oral and Maxillofacial Surgery

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Case report

Botryo im

Kazumas imaDivision of Firs eikai1-1 Keyaki-dai

a r t i c l

Article history:Received 7 JulReceived in re21 SeptemberAccepted 25 OAvailable onlin

Keywords:Botryoid odonCytokeratin prGlycogen

enicimm

iumK19, a-immic ke

th glyion.ation

1. Introdu

Botryoidalveolar bothe initial report of two BOCs by Weathers and Waldron [1,2], morethan 67 cases have been reported in the English literature [3]. Thelesion is generally considered to represent a variant of the lateralperiodontal cyst (LPC) [48] possibly the result of cystic degen-eration and subsequent fusion of adjacent foci of dental laminarests [5]. Aas LPC, is thbeen reporument furthlesion. In thwith histocthe cyst-lin

2. Case rep

A 59-yeMeikai Univin the left atomatic swexaminatiodelimited, mthe left ma

CorresponE-mail add

cencapicwas

entriepithelium (Fig. 2a). The brous connective tissue wall was rela-tively free of inammatory cells. The epitheliumexhibited localizedplaques with many clear cells containing centrally placed ovoidnuclei (Fig. 2b). The supercial layer of the epithelium showedcuboidal to columnar cells that were sometimes ciliated. These

0915-6992/$ doi:10.1016/j.lthough conservative enucleation of the BOC, as welle treatment of choice, a signicant recurrence rate hasted for BOC [915]. It is, therefore, of interest to doc-er characteristics of the cyst-lining epithelium of thee present study, we report an additional case of BOChemical and immunohistochemical characteristics ofing epithelium.

ort

ar-old female patient was referred to the hospital ofersity School of Dentistry for evaluation of a swellingnterior mandible. She had been aware of the asymp-elling during the past two years. At the time of dentaln, panoramic and dental radiographs revealed a well-

ulti-locular radiolucent lesion, between the roots ofndibular lateral incisor and canine (Fig. 1). The cystic

ding author. Tel.: +81 49 279 2757; fax: +81 49 279 2757.ress: kazu-mori@dent.meikai.ac.jp (K. Mori).

epithelial elements showed a diastase digestible PAS reaction, indi-cating the presence of glycogen (Fig. 3). On the basis of clinical andhistopathological ndings, the lesion interpreted as a BOC.

Positive immunoreactivities were obtained for CKs 10/13, 14and 19 (Fig. 4ac); there was expression on CK 10/13 and CK19 inthe spinous and surface layers, CK14 in the basal cell layer. CK 18weakly expressed only in the supercial layer of the epithelium(not shown). In addition to the cytokeratin proles, immunos-taining for proliferating cell nuclear antigen (PCNA) was alsoperformed. PCNA-reactive cellswere insignicant in theepitheliumof BOC (Fig. 5a) as compared to that of the odontogenic keratocyst(currently termed keratocystic odontogenic tumor; 2005, WHO)(Fig. 5b) as a positive control section, in which an abundance of theimmunoreactive cells could be seen.

3. Discussion

The BOC was originally described by Weathers and Waldron in[1]. The term was based on the clinical appearance that resembleda bunch of grapes. The BOC has a distinct proclivity for occurrencein the mandible anterior to the rst molar [16]. Radiographically,

see front matter 2010 Asian Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd.All rights reserved.ajoms.2010.10.002id odontogenic cyst: A case report with

a Mori , Nozomi Tamura, Nobuaki Tamura, Jun Sht Oral and Maxillofacial Surgery, Department of Diagnosis and Therapeutics Sciences, M, Sakado, Saitama, 350-0283, Japan

e i n f o

y 2010vised form2010ctober 2010e 8 December 2010

togenic cystole

a b s t r a c t

We present a case of botryoid odontogfemale patient. The histochemical andThe nature of the cyst-lining epithelexpression of CK 10/13, CK14, and Cenamel organ. An insignicant PCNAcompared with that of the odontogenPAS-reactive material (consistent wilamina are a possible origin of the les

2010 Asian Associ

ction

odontogenic cyst (BOC) is a polycystic lesion in thene with or without proximity to a root of tooth. Since

radioluto thebiopsymulticm/locate /a joms

munohistochemical aspects

daUniversity School of Dentistry,

cyst, affecting the anterior mandible in a 59-year-old Japaneseunohistochemical characteristics of the lesion are described.

was suggestive of an odontogenic origin as revealed by thell of which have been reported to be present in the humanunoreaction was observed in the cyst-lining epithelium asratocyst. Furthermore, the appearance of diastase digestible,cogen) was evaluated that prefunctional cells of the dental

of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd.All rights reserved.

y occupied the lateral aspects of the teeth and extendedal region with separation of the roots. An excisionalperformed. Histopathologically, the lesion showed a

c cystic conguration lined by thin layer of squamous

32 K. Mori et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 3134

Fig. 1. Radiographs showing a multilocular radiolucency between the roots of themandibular canine and lateral incisor. The cystic lesion extends from the lateralaspects of the roots of teeth over the apical region with separation of the roots.

it is often multilocular and larger than the typical LPC, and oftenextends into the periapical regions of the related teeth. These twolesions share some histologic similarities; they contain character-istic thickened epithelial plaques or clear cell nests in the epitheliallining. Due to the similarity in histologic features and site of occur-rence, the BOC has been considered a variant of the LPC [49].

The signicance of separating the BOC from LPC is based on thesize and gross appearance of the former; the BOC is more expan-sive than the LPC because of its multicentric nature. The higherrecurrence rate of BOC is not because of the cell growth activity,but because of difculty in compete surgical removal of a multiloc-ular lesion [17]. Therefore, an extended postsurgical follow-up isrecommended clinically.

Fig. 2. Low-posquamoid epitthickenings an(b).wer photomicrograph demonstrates the polycystic nature with thinhelial lining (a). Cyst-lined epithelium exhibiting focal plaque-liked clear cells with cuboidal to columnar cells at the supercial layer

Fig. 3. PAS reaof glycogen in

Becausecells, BOC sodontogeniHowever, thmay lie at odoes not dereported inctions with (a) or without (b) diastase digestion reveal the presencethe cyst-lyning epithelium.

of the presence of mucous cells and surface columnarhows some microscopic similarities to the glandularc cyst (GOC) or sialo-odontogenic cyst (SOC) [1820].ese lesions were felt to be best classied as a BOC. They

pposite endsof a spectrum.Thepresenceofmucous cellstract from an odontogenic origin, this feature has beena variety of odontogenic cysts such as the dentigerous

K. Mori et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 3134 33

Fig. 4. CK immthe spinous ancell layer.

cyst as a mhistochemihistologic v

ImmunoHeikinheimpresence ofTheseauthoactions of Cductal andof origin of Bbe found. Inenamel orgmediate la13 and 19near the surpositive stathe cyst-linunoreactivities of CK 10/13 (a) and CK19 (c) show positive rections ind surface layers, while CK14 (b) reveals a reactivity only in the basal

etaplastic phenomenon [21]. Furthermore, immuno-cal studies [22,23] have suggested that the GOC is aariant of BOC.histochemical analysis on the BOC was performed byo et al. [13]; an odontogenic origin is supported theCK 19 immunoreactivity of the cyst-lining epithelium.rs alsoobservedaheterogeneouslypositive immunore-K 18, which has been recognized as a marker of simpleglandular epithelia. They stressed the odontogenic cellOC inwhich patchy distribution of CKs 13 and 16 couldthis regard, CKs 7, 13, 14 and 19 are present in human

an [24]. Crivelini et al. [25] concluded that typical inter-ment of odontogenic epithelium is CK 14, and that CKsappear in squamous differentiation or epithelial cellsface epithelium. In the present study, we also observedining for CKs 10/13, 14 and 19 in the respective layers ofing epithelium. Considering these evidences, it is appar-

Fig. 5. PCNApositive contr

ent that thebeen demo

Severalglycogen, ddigestible, mstudy, glycothe epithelsome of thShear [7] ststrable in th[26] observepithelium

In a reviwhich demstudy, wein the cyst-genic keratepithelial cbiologicallyfor the limiwell), compmer arisesthe latter prpossessingcould be cit

Acknowled

We thanolina, for hiDr. Y. TajimSchool of Dimmunoreactivity in the BOC (a) and OKC (b), the latter used as aol lesion.

epithelial cells of BOC are of odontogenic origin, as hasnstrated by earlier investigators [5,7].investigators have identied the sporadic presence of

etected as periodic acid-Schiff (PAS) positive, diastaseaterial in the lining epithelium [16,26]. In the presentgen was detected in the cyst-lining epithelium and in

ial plaques, but not always in the clear cells, althoughese cells were PAS positive. In this respect, Altini andated that glycogen is by no means consistently demon-e clear cells and is not conned to them. Redman et al.ed no clear cells in their case, while the cyst-liningcontained an amount of glycogen.ew of the English literature, no reports could be foundonstrated the proliferative activity of the BOC. In thiscould detect an insignicant PCNA immunoreactivitylining epithelium in comparison with that of odonto-ocyst. It can therefore be speculated that the cyst-liningells, in which an amount of glycogen was detected, areinactive. Altini and Shear [7] suggested that the reasonted growth potential of the LPC (and that of the BOC asared with the odontogenic keratocyst is that the for-

from prefunctional cells of the dental lamina, whereasesumably arises from that part of the dental lamina stillmarked growth potential. The results of present studyed in support of this evidence.

gements

k Dr. Brad W. Neville, Medical University of South Car-s provisional review of the manuscript. We also thanka, Department of Oral Pathology, Meikai University

entistry, for preparing this manuscript.

34 K. Mori et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 3134

This work was supported in part by Miyata Memorial Fund ofthe Meikai University.

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Botryoid odontogenic cyst: A case report with immunohistochemical aspectsIntroductionCase reportDiscussionAcknowledgementsReferences