Unusual Cystic Variant of Calcifying Epithelial Odontogenic Tumor

Abstract

Calcifying epithelial odontogenic tumor (CEOT) is a rare benign odontogenic neoplasm, which is exclusively epithelial in its tissue of origin. Many cases of CEOTs are associated with impacted tooth and simulate dentigerous cyst radiographically. The histologic features of CEOT are unique; however, among its various histologic subtypes, the cystic variant is a rare and less well-understood entity. Our report elucidates a cystic variant of CEOT in the maxilla of a 16-year-old male that presents clinical and radiologic findings conscientious to dentigerous cyst; but histopathological diagnosis came out to be a gold standard in identifying this rare tumor. This case report describes the clinicopathologic features of this rare entity, highlighting the histomorphological findings along with reviewing other reported cases.

Keywords: Calcifying epithelial odontogenic tumor; Cystic variant; Maxilla; Odontogenic.

Figure 1

a; Clinical extra- oral and b; intra- oral view

Figure 2

a: Panoramic radiography revealing unilocular radiolucency above the root apices of left maxillary second premolar and first molar involving unerupted left maxillary second molar in center, b: Computed tomography (CT) scan shows a well- defined unilocular radiolucency in the left maxillary sinus, with impacted tooth and few flecks of calcified deposits

Figure 3

Intra- operative picture of surgical approach

Figure 4

one large cystic specimen attached at the cemento- enamel junction of the impacted tooth and other separated bit

Figure 5

Cystic cavity lined by odontogenic epithelium of varying thickness composed of polyhedral cells with a distinct outline and centrally placed hyperchromatic nuclei (H&E; 10x, 20x, 40x)

Figure 6

Sheet of polyhedral epithelial cells interspersed by calcifications and homogeneous eosinophilic amyloid-like material stained positive for Congo red and exhibiting minimal apple-green birefringence under polarizing microscope (H&E; 10x, 20x and Congo red;10x, 40x).