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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
CASE REPORT Table of Contents   
Year : 2006  |  Volume : 10  |  Issue : 1  |  Page : 17-19
 

Peripheral clear cell variant of calcifying epithelial odontogenic tumour


Department of Oral Pathology and Microbiology, Government Dental College and Hospital, GHATI, Aurangabad-431 001, India

Correspondence Address:
J V Tupkari
Department of Oral Pathology and Microbiology, Government Dental College and Hospital, GHATI, Aurangabad-431 001
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-029X.37790

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   Abstract 

The calcifying epithelial odontogenic tumour (CEOT) was first described as an entity by Pindborg in 1955. It is classified as an uncommon, benign, odontogenic neoplasm that is exclusively epithelial in origin. CEOT represents less than 1 % of all odontogenic tumours. Of all reported CEOTs, peripheral tumours account for 5-6%, among which clear cell variants are still rarer. This article presents the clinical, radiological and microscopic features of a peripheral clear cell variant of CEOT occurring in the gingiva of the anterior mandible of a 22-year-old female.


Keywords: Calcifying epithelial odontogenic tumour, clear cell.


How to cite this article:
Shetty S, Tupkari J V, Barpande S R. Peripheral clear cell variant of calcifying epithelial odontogenic tumour. J Oral Maxillofac Pathol 2006;10:17-9

How to cite this URL:
Shetty S, Tupkari J V, Barpande S R. Peripheral clear cell variant of calcifying epithelial odontogenic tumour. J Oral Maxillofac Pathol [serial online] 2006 [cited 2019 Aug 23];10:17-9. Available from: http://www.jomfp.in/text.asp?2006/10/1/17/37790



   Introduction Top


The calcifying epithelial odontogenic tumour (CEOT) is an uncommon lesion that accounts for less than 1% of all odontogenic tumours [1]. It was first described by Pindborg [2], and is referred to as Pindborg's tumour [3]. Fewer than 200 cases have been reported till date. By far the most common CEOT location is intraosseous, but it also may be extraosseous. The extraosseous or peripheral variant was first observed by Pindborg in 1966, and it represents approximately 6% of the total cases of CEOT [3].

Under microscopic examination, the classical pattern of CEOT is a combination of cords, nests, or sheets of polyhedral epithelial cells, amyloid-like deposits, and concentric calcified structures (Liesegang's rings). The clear cell variant of CEOT (CCEOT) was first described by Abrams and Howell. It is characterized by polyhedral epithelial cells alternating with huge epithelial cells with a clear, foamy cytoplasm; distinct cell borders; moderate variation in nuclear size; some vacuolated nuclei; and no extreme hyperchromatism or bizarre nuclei [3]. Sixteen cases of CCEOT have been described in the literature, and eight of them represent the peripheral variant. The mean age of the patients with peripheral CCEOT is 35.13 years (range, 16 to 64 years) at the time of diagnosis, and the male to female ratio is identical (M/F =1:1). All cases of peripheral CCEOT were located in the gingiva, with the mandible more commonly affected than the maxilla (mandible: maxilla= 5:3) [3],[4].

The purpose of this article is to present the clinical, radiological and microscopic characteristics of a peripheral CCEOT occurring in the gingiva of the anterior mandible of a 22-year-old female. This case adds to the very few cases of peripheral CCEOT reported in the literature.


   Case Report Top


A 22-year-old female reported with the complaint of a growth in the anterior region of lower jaw since four years. There were no other associated complaints. Intraoral examination revealed the presence of a solitary, sessile, firm well-circumscribed, roughly oval growth between 32 and 33. It measured approximately 2cm x 1cm x 1cm. It was pinkish red in colour with foci of brownish discolouration at places. The surface was smooth with stippling present at superior border. The growth was confined to the gingiva, extending apicocoronally from mucogingival junction to near the incisal edges of 32 and 33. Pathologic migration of 33, 34, 35 and 36 was observed [Figure - 1].

Intraoral periapical radiograph revealed mesial migration of 33, 34 and distal migration of 34, 35. Superficial erosion of alveolar bone between 32 and 33 was noticed [Figure - 2]. The lesion was treated by surgical excision. Post surgical healing was uneventful. Haematoxylin and eosin stained sections of the excised mass showed the presence of a covering of parakeratinized stratified squamous epithelium. Underlying connective tissue showed polyhedral, epithelial cells with dense eosinophilic cytoplasm, hyperchromatic nuclei, and prominent intercellular bridges admixed with regions of cells exhibiting a clear vacuolated cytoplasm [Figure - 3],[Figure - 4] and [Figure - 5]. Foci of calcification were also seen. Areas of eosinophilic, homogeneous material represented amyloid deposition as revealed by the 'apple-green birefringence' of the Congo red stain with polarized light [Figure - 6]. The tumour cells were immunonegative for S-100 protein.

Based on clinical, radiological, histopathological, and immunohistochemical findings, the case was diagnosed as a peripheral clear cell variant of CEOT.


   Discussion Top


The peripheral CCEOT is said to arise from epithelial rests of dental lamina or basal cells of the gingival surface epithelium [5]. It has been shown both histochemically and in electron microscopy, that the clear cells in CCEOT contain glycogen [7]. In CCEOT, clear cell areas may be a minor focal component or may constitute the majority of the tumour tissue [5]. In the present case, clear cell areas constituted the major portion of the tumour mass. The diagnosis of CCEOT is usually based on the finding of some areas of typical polyhedral epithelial cells within the tumour [6]. The ratio of peripheral to central lesions in CEOT is 1:24, whereas the same ratio in CCEOT is 1:1.56, excluding the case of peripheral CCEOT reported by Mesquita et al (2003). This indicates that CCEOT has a greater tendency to develop on the gingiva [3],[4]. Peripheral CCEOT has a preference for the anterior to bicuspid areas (six out of eight reported cases) [3],[4]. In peripheral CEOT, calcification is minimal or may be totally absent [3]. Calcified material in small quantity was observed in the present case, but it was not in the form of Liesegang's rings.

Although the gingiva is not a common location for salivary gland neoplasms, microscopically clear cell variants of salivary gland tumours should be included in the differential diagnosis of peripheral CCEOT [8]. In the present case, the presence of amyloid, calcifications, and absence of S-100 protein expression, ruled out the diagnosis of clear cell salivary gland tumours. Hicks et al suggested that the presence of clear cells in CEOT may confer a more aggressive behavior to the lesion [4]. In the eight cases of peripheral CCEOT reported in the literature, the presence of clear cell component was not associated with aggressiveness. However, too few cases of peripheral CCEOT have been described to date to reach a clear conclusion regarding the impact of the clear-cell population on the tumours biologic activity.

 
   References Top

1.Neville BW, Damm DD, Allen CM, Bouquot JE (2004): Oral and maxillofacial pathology, (2nd ed.), Elsevier, New Delhi.  Back to cited text no. 1    
2.Shafer WO; Hine MK, Levy BM (1983): A textbook of oral pathology, (4'h ed.), W.B.Saunders company, Philadelphia.  Back to cited text no. 2    
3.Mesquita RA, Lotufo MA, Sugaya NN, Araujo NS, Araujo VC (2003): Peripheral clear cell variant of calcifying epithelial odontogenic tumour: report of a case and immunohistochemical investigation, Oral Surg Oral Med Oral Pathol, 95:198-204.  Back to cited text no. 3    
4.Anavi Y, Kaplan I, Citir M, Calderon S (2003): Clear-cell variant of calcifying epithelial odontogenic tumour: clinical and radiological characteristics. Oral Surg Oral Med Oral Pathol, 95:332-9.  Back to cited text no. 4    
5.Orsini Q Favia Ci Piatelli A (2000): Peripheral clear cell calcifying epithelial odontogenic tumour. Report of a case, J Periodontol, 71:1177-1180.  Back to cited text no. 5    
6.Cawson RA, Binnie WH, Speight PM, Barrett AW, Wright JM (1998): Lucas' Pathology of tumours of the oral tissues, (5d' ed.), Churchill Livingstone, London.  Back to cited text no. 6    
7.Philipsen HP & Reichart PA (2000): Calcifying epithelial odontogenic tumour: biological profile based on 181 cases from the literature, Oral Oncol 36:17-26.  Back to cited text no. 7    
8.Houston GD & Fowler CB (1997): Extraosseous calcifying epithelial odontogenic tumour: report of two cases and review of the literature, Oral Surg Oral Med Oral Pathol 83:577-83.  Back to cited text no. 8    


    Figures

  [Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5], [Figure - 6]



 

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    Abstract
    Introduction
    Case Report
    Discussion
    References
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