Hybrid desmoplastic ameloblastoma: A case report of rare entity and review of literature

Abstract

Ameloblastomas are one of the common odontogenic tumors of the oral cavity. They usually present with unilocular or multilocular radiolucency associated with or without impacted tooth. Among the variants of ameloblastomas, desmoplastic ameloblastoma (DA) casts a mixed radiolucent/radiopaque shadow on X-ray and radiograph seldom suggests a diagnosis of ameloblastoma. DA is a rare entity that exhibits important differences in anatomic distribution, histologic appearance, radiographic findings when compared to other variants of ameloblastoma. This fact has been considered by World Health Organization (WHO) and now it has been included as a separate entity in WHO classification (2005) of odontogenic tumors. About 145 cases of DA have been reported worldwide, but cases reported in India are very few. We report a case of a hybrid variety of DA in a female patient in the anterior maxillary region between 12 and 13 as painless hard swelling, showing mixed radiolucent/radiopaque shadow on radiographic examination. Histopathology revealed odontogenic epithelium in the form of follicles, typical of solid/multicystic ameloblastoma, with cystic degeneration and squamous metaplasia at places and elsewhere there were odontogenic islands compressed by dense fibrocellular stroma suggestive of desmoplasia. Also seen was osseous metaplasia. We have also done a detailed review of literature concerning the presentation of DA.