Unicystic ameloblastoma is a rare variant of ameloblastoma. We report a case of Unicystic ameloblastoma in a 29 year-old female with a pain and swelling in the left mandibular posterior region. Fine needle aspiration yielded no fluid. Periapical, panoramic and computer tomography scans showed well defined radiolucency present in relation to 36 and 37. Unicystic refers to those cystic lesions that show clinical, radiographic, or gross features of a cyst, but on histologic examination show a typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumor growth. Enucleation and Tumor resection was performed for the treatment. Our case report is an attempt to help the dental community in developing familiarity with the clinical presentation and at the same time advocating to develop a high index of suspicion in recognizing such cases.

Keywords: Unicystic Ameloblastoma; Odontogenic tumor; Histopathology.