Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 31–34
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Asian Journal of Oral and Maxillofacial Surgery
j o u r n a l h o m e p a g e :w w w . e l s e v i e r . c o m / l o c a t e / a j o m s
Botryoid odontogenic cyst: A case report with immunohistochemical aspects
, Nozomi Tamura, Nobuaki Tamura, Jun Shimada
Division of First Oral and Maxillofacial Surgery, Department of Diagnosis and Therapeutics Sciences, Meikai University School of Dentistry, 1-1 Keyaki-dai, Sakado, Saitama, 350-0283, Japan
a r t i c l e i n f o
Received 7 July 2010 Received in revised form 21 September 2010 Accepted 25 October 2010 Available online 8 December 2010
Botryoid odontogenic cyst Cytokeratin proﬁle Glycogen
a b s t r a c t
We present a case of botryoid odontogenic cyst, affecting the anterior mandible in a 59-year-old Japanese female patient. The histochemical and immunohistochemical characteristics of the lesion are described.
The nature of the cyst-lining epithelium was suggestive of an odontogenic origin as revealed by the expression of CK 10/13, CK14, and CK19, all of which have been reported to be present in the human enamel organ. An insigniﬁcant PCNA-immunoreaction was observed in the cyst-lining epithelium as compared with that of the odontogenic keratocyst. Furthermore, the appearance of diastase digestible, PAS-reactive material (consistent with glycogen) was evaluated that prefunctional cells of the dental lamina are a possible origin of the lesion.
© 2010 Asian Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd.
All rights reserved.
Botryoid odontogenic cyst (BOC) is a polycystic lesion in the alveolar bone with or without proximity to a root of tooth. Since the initial report of two BOCs by Weathers and Waldron[1,2], more than 67 cases have been reported in the English literature. The lesion is generally considered to represent a variant of the lateral periodontal cyst (LPC) [4–8]possibly the result of cystic degen- eration and subsequent fusion of adjacent foci of dental lamina rests. Although conservative enucleation of the BOC, as well as LPC, is the treatment of choice, a signiﬁcant recurrence rate has been reported for BOC[9–15]. It is, therefore, of interest to doc- ument further characteristics of the cyst-lining epithelium of the lesion. In the present study, we report an additional case of BOC with histochemical and immunohistochemical characteristics of the cyst-lining epithelium.
2. Case report
A 59-year-old female patient was referred to the hospital of Meikai University School of Dentistry for evaluation of a swelling in the left anterior mandible. She had been aware of the asymp- tomatic swelling during the past two years. At the time of dental examination, panoramic and dental radiographs revealed a well- delimited, multi-locular radiolucent lesion, between the roots of the left mandibular lateral incisor and canine (Fig. 1). The cystic
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radiolucency occupied the lateral aspects of the teeth and extended to the apical region with separation of the roots. An excisional biopsy was performed. Histopathologically, the lesion showed a multicentric cystic conﬁguration lined by thin layer of squamous epithelium (Fig. 2a). The ﬁbrous connective tissue wall was rela- tively free of inﬂammatory cells. The epithelium exhibited localized plaques with many clear cells containing centrally placed ovoid nuclei (Fig. 2b). The superﬁcial layer of the epithelium showed cuboidal to columnar cells that were sometimes ciliated. These epithelial elements showed a diastase digestible PAS reaction, indi- cating the presence of glycogen (Fig. 3). On the basis of clinical and histopathological ﬁndings, the lesion interpreted as a BOC.
Positive immunoreactivities were obtained for CKs 10/13, 14 and 19 (Fig. 4a–c); there was expression on CK 10/13 and CK19 in the spinous and surface layers, CK14 in the basal cell layer. CK 18 weakly expressed only in the superﬁcial layer of the epithelium (not shown). In addition to the cytokeratin proﬁles, immunos- taining for proliferating cell nuclear antigen (PCNA) was also performed. PCNA-reactive cells were insigniﬁcant in the epithelium of 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 the immunoreactive cells could be seen.
The BOC was originally described by Weathers and Waldron in . The term was based on the clinical appearance that resembled a bunch of grapes. The BOC has a distinct proclivity for occurrence in the mandible anterior to the ﬁrst molar. Radiographically, 0915-6992/$ – see front matter © 2010 Asian Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd.All rights reserved.
32 K. Mori et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 31–34
Fig. 1. Radiographs showing a multilocular radiolucency between the roots of the mandibular canine and lateral incisor. The cystic lesion extends from the lateral aspects 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 often extends into the periapical regions of the related teeth. These two lesions share some histologic similarities; they contain character- istic thickened epithelial plaques or clear cell nests in the epithelial lining. Due to the similarity in histologic features and site of occur- rence, the BOC has been considered a variant of the LPC[4–9].
The signiﬁcance of separating the BOC from LPC is based on the size and gross appearance of the former; the BOC is more expan- sive than the LPC because of its multicentric nature. The higher recurrence rate of BOC is not because of the cell growth activity, but because of difﬁculty in compete surgical removal of a multiloc- ular lesion. Therefore, an extended postsurgical follow-up is recommended clinically.
Fig. 2. Low-power photomicrograph demonstrates the polycystic nature with thin squamoid epithelial lining (a). Cyst-lined epithelium exhibiting focal plaque-like thickenings and clear cells with cuboidal to columnar cells at the superﬁcial layer (b).
Fig. 3. PAS reactions with (a) or without (b) diastase digestion reveal the presence of glycogen in the cyst-lyning epithelium.
Because of the presence of mucous cells and surface columnar cells, BOC shows some microscopic similarities to the glandular odontogenic cyst (GOC) or sialo-odontogenic cyst (SOC)[18–20].
However, these lesions were felt to be best classiﬁed as a BOC. They may lie at opposite ends of a spectrum. The presence of mucous cells does not detract from an odontogenic origin, this feature has been reported in a variety of odontogenic cysts such as the dentigerous
K. Mori et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 31–34 33
Fig. 4. CK immunoreactivities of CK 10/13 (a) and CK19 (c) show positive rections in the spinous and surface layers, while CK14 (b) reveals a reactivity only in the basal cell layer.
cyst as a metaplastic phenomenon . Furthermore, immuno- histochemical studies [22,23]have suggested that the GOC is a histologic variant of BOC.
Immunohistochemical analysis on the BOC was performed by Heikinheimo et al. ; an odontogenic origin is supported the presence of CK 19 immunoreactivity of the cyst-lining epithelium.
These authors also observed a heterogeneously positive immunore- actions of CK 18, which has been recognized as a marker of simple ductal and glandular epithelia. They stressed the odontogenic cell of origin of BOC in which patchy distribution of CKs 13 and 16 could be found. In this regard, CKs 7, 13, 14 and 19 are present in human enamel organ. Crivelini et al.concluded that typical inter- mediate ﬁlament of odontogenic epithelium is CK 14, and that CKs 13 and 19 appear in squamous differentiation or epithelial cells near the surface epithelium. In the present study, we also observed positive staining for CKs 10/13, 14 and 19 in the respective layers of the cyst-lining epithelium. Considering these evidences, it is appar-
Fig. 5. PCNA immunoreactivity in the BOC (a) and OKC (b), the latter used as a positive control lesion.
ent that the epithelial cells of BOC are of odontogenic origin, as has been demonstrated by earlier investigators[5,7].
Several investigators have identiﬁed the sporadic presence of glycogen, detected as periodic acid-Schiff (PAS) positive, diastase digestible, material in the lining epithelium[16,26]. In the present study, glycogen was detected in the cyst-lining epithelium and in the epithelial plaques, but not always in the clear cells, although some of these cells were PAS positive. In this respect, Altini and Shearstated that glycogen is by no means consistently demon- strable in the clear cells and is not conﬁned to them. Redman et al.
 observed no clear cells in their case, while the cyst-lining epithelium contained an amount of glycogen.
In a review of the English literature, no reports could be found which demonstrated the proliferative activity of the BOC. In this study, we could detect an insigniﬁcant PCNA immunoreactivity in the cyst-lining epithelium in comparison with that of odonto- genic keratocyst. It can therefore be speculated that the cyst-lining epithelial cells, in which an amount of glycogen was detected, are biologically inactive. Altini and Shearsuggested that the reason for the limited growth potential of the LPC (and that of the BOC as well), compared with the odontogenic keratocyst is that the for- mer arises from prefunctional cells of the dental lamina, whereas the latter presumably arises from that part of the dental lamina still possessing marked growth potential. The results of present study could be cited in support of this evidence.
We thank Dr. Brad W. Neville, Medical University of South Car- olina, for his provisional review of the manuscript. We also thank Dr. Y. Tajima, Department of Oral Pathology, Meikai University School of Dentistry, for preparing this manuscript.
34 K. Mori et al. / Asian Journal of Oral and Maxillofacial Surgery 23 (2011) 31–34
This work was supported in part by Miyata Memorial Fund of the Meikai University.
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