Aggressive presentation of ameloblastic fibro-odontoma: a clinical-pathological enigma

Abstract

Ameloblastic fibro-odontoma (AFO) is a rare, gnathic, benign, mixed odontogenic tumor that commonly presents in the first or second decade of life as a unilocular and rarely multilocular radiolucency with variable amounts of calcified material. Tumor progression is typically indolent and generally-accepted treatment is surgical enucleation and curettage. This case report describes an atypical presentation in a 14-year-old male with a multilocular, aggressive AFO requiring hemimandibulectomy with immediate osseous and dental, “Jaw in a Day” (JIAD), reconstruction. This report highlights the debate regarding whether AFO is a true neoplasm or an early-stage hamartoma in the continuum of complex odontoma formation. Regardless of the pathogenesis, maxillofacial surgeons and pathologists should be cognizant of the potential for AFO to develop locally aggressive behavior with considerable morbidity.