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Year : 2008  |  Volume : 51  |  Issue : 4  |  Page : 576-577
A rare variant of ameloblastoma associated with a glandular odontogenic cyst


Department of Oral Pathology and Microbiology, Bapuji Dental College and Hospital Davangere, Karnataka, India

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How to cite this article:
Kumaraswamy Naik L R, Agarwal R, Amberkar VS, Ahmed Mujib B R. A rare variant of ameloblastoma associated with a glandular odontogenic cyst. Indian J Pathol Microbiol 2008;51:576-7

How to cite this URL:
Kumaraswamy Naik L R, Agarwal R, Amberkar VS, Ahmed Mujib B R. A rare variant of ameloblastoma associated with a glandular odontogenic cyst. Indian J Pathol Microbiol [serial online] 2008 [cited 2020 Feb 23];51:576-7. Available from: http://www.ijpmonline.org/text.asp?2008/51/4/576/43775


Ameloblastoma has been extensively reviewed in literature whereas glandular odontogenic cysts (GOC) have only recently been described by Gardner, et al. in 1988. [1] GOC is a rare developmental cyst of the jaw, reported in just over 50 cases in literature. [1] The case report describes an intra-bony lesion in the mandible of a 14-year-old boy that though clinically resembled a dentigerous cyst, histopathology revealed to be a calcifying ameloblastoma with a glandular odontogenic cyst.

A 14-year-old boy presented with the chief complaint of pain and a bony hard swelling in the right lower third of his face since the past 8 months. Intra-orally, the swelling was extended from the canine to the retro molar area obliterating the buccal vestibule. The lower right second premolar and first molar were non vital. A radiographic examination revealed a well-circumscribed radiolucenct lesion in the right mandible. The radiolucency was surrounded by well corticated borders and there was a presence of root resorption. The right second molar was inferiorly displaced and the third molar tooth bud was displaced to the ramus region.

Aspiration from the lesion revealed blood tinged fluid and total protein content of 5 gm per cent. Cholesterol crystals were seen in unstained aspirate. With the working diagnosis of dentigerous cyst, the lesion was enucleated under general anesthesia.

An examination of the gross specimen revealed an intact cystic lesion encircling the neck of the mandibular second molar. Multiple sections from the specimen showed cystic lesions with dense fibrous connective tissue capsule. The lining was variable, with areas of reduced enamel epithelium (REE), which in a few areas, was progressing to ameloblastoma [Figure 1]. A few areas of this ameloblastoma lining showed aberrant keratinization and calcifications in the suprabasal cells. These calcifications were in the form of spherical nodules and in some areas they were in a concentric lamellar pattern [Figure 2]. A few areas of calcification were also noted in the connective tissue capsule along with the epithelium. Clear cell formation was also evident in a few areas. Some of the proliferating ameloblastoma follicles were noted within the fibrous capsule [Figure 3]. Many areas of the lining epithelium revealed features of GOC [Figure 4] in the form of a stratified squamous epithelium with superficial cuboidal to columnar cells. Cilia were present in a few of these superficial layers. Pseudo-stratified ciliated columnar epithelial lining was also evident. Epithelial plaque was observed in a few areas. Intraepithelial clefting was also noted. Fibrous capsule appeared non inflamed and extensively hyalinized. Periodic acid schiff (PAS), Congo red, and mucicarmine were negative. Depending upon the clinical, radiological, and histological features, a final diagnosis of calcifying unicystic ameloblastoma associated with glandular odontogenic cyst was made. The patient has been on follow-up for the past 1 year.

GOC has a wide clinicopathologic spectrum. Hence the provisional diagnosis, based on clinical and radiological examination, may vary. [1] The present case showed an impacted tooth with cystic attachment at the neck (Cementoenamel junction) and histopathologically REE lining fulfilling the definition of dentigerous cyst. [2] The lining epithelium also showed features that were characteristically described for GOC though mucicarmine and PAS were negative. It is not necessary to find mucous pool in the cyst lining, and GOC has been diagnosed based on other characteristic features in the absence of mucous pool. An important diagnostic feature is the surface layer of cuboidal cells, which was present in this case. [3]

In the present case, features of unicystic ameloblastoma (group 3a type) were observed with a few areas showing mural proliferation of ameloblastoma follicles suggesting the origin of ameloblastoma was from the cystic lining. [2] Similarly, the origin of ameloblastoma from the lining of the dentigerous cyst has also been reported in literature. [2] Kramer IRH, Pindborg JJ, Shear M identified some variants of ameloblastomas showing dystrophic calcifications within the epithelium similar to this case. [4] Also, presence of cilia as seen in the present case is not ordinarily seen in ameloblastoma, although the tumor cells have a high ability to undergo various forms of metaplasia. [5] The present lesion is differentiated from the calcifying epithelial cyst because of the absence of the characteristic and distinguishing features of the ghost cell formation. [4]

GOC radiographically presenting as a dentigerous cyst similar to this case has been reported previously three times. [1] There are only two reported cases of the simultaneous occurrence of GOC and ameloblastoma. [5] The association of GOC with conventional ameloblastoma has been reported by Hisatomi, et al. [5] but GOC with unicystic ameloblastoma has not been reported in literature.

Hence, it is thought that this could be the first case report of a dentigerous cyst progressing into a glandular odontogenic cyst and calcifying unicystic ameloblastoma. It is of less possibility that the present lesion is the result of a collision of three different lesions. All the pathologic slides were subjected to peer review and no sign of muco-epidermoid carcinoma was found in any of the sections examined.

In conclusion, the present case report emphasizes the pleuripropensity of the odontogenic epithelium especially the REE of the dentigerous cyst lining. The case clinically and radiographically presented as a less aggressive dentigerous cyst, while histopathology revealed a hybrid aggressive lesion. Hence, multiple sections of the specimen should be examined thoroughly to exclude the possibility of hybrid lesions especially when associated with an impacted tooth. Further studies are required to study the clinical and biological behavior of these hybrid lesions.

 
   References Top

1.Kasaboglu O, Basal Z, Usubutun A. Glandular odontogenic cyst presenting as a dentigerous cyst: a case report, J Oral Maxillofac Surg 2006;64:731-3.  Back to cited text no. 1    
2.Shear M: Dentigerous (folliculr) cyst. In: Wright, editor. Cysts of the oral regions. 3 rd ed. Butterworth Heinmann Ltd; 1992. p. 75-98.   Back to cited text no. 2    
3.Gardner DG, Kessler HP, Morency R, Schaffner DL. The glandular odontogenic cyst: An apparent entity. J Oral Pathol 1988;17:359-66.  Back to cited text no. 3  [PUBMED]  
4.Kramer IRH, Pindborg JJ, Shear M. Neoplasms and other related to the odontogenic apparatus. In: hystological typing of odontogenic tumours. WHO International Histological classification of tumours. 2 nd ed.. Springer- Verlag; 1992. p. 11-21.  Back to cited text no. 4    
5.Hisatomi M, Asaumi J, Konouchi H, Yanagi Y, Kishi K. A case of glandular odontogenic cyst associated with ameloblastoma: Correlation of diagnostic imaging with histopathological features. Dentomaxillofac Radiol 2000;29:249-53.  Back to cited text no. 5  [PUBMED]  [FULLTEXT]

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Correspondence Address:
L R Kumaraswamy Naik
Department of Oral Pathology and Microbiology, Bapuji Dental College and Hospital, Davangere 577 004, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.43775

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

This article has been cited by
1 Glandular odontogenic cyst Literature review and report of a paediatric case
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Journal of Oral Biology and Craniofacial Research. 2015; 5(3): 219
[Pubmed] | [DOI]



 

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