• Sushil Rayamajhi, Sunita Shrestha, Samata Shakya, Sagar Bhandari, Anu Radha Twayana, and Kopila Shahi.
• Department of Medicine, Swacon International Hospital, Rudramati Marg, Kathmandu, Nepal.
• JNMA J Nepal Med Assoc. 2022 Jul 1; 60 (251): 657660657-660.

UnlabelledAmeloblastomas of jaws are benign odontogenic tumors of epithelial origin with four clinical variants: solid multicystic type, unicystic type, desmoplastic type, and extraosseous type. The incidence rate of ameloblastoma is 0.92 per million person-years. Unicystic ameloblastoma refers to those cystic lesions that show clinical and radiologic characteristics of an odontogenic cyst but shows a typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumor proliferation on histological examination. Here is a unique case of unicystic ameloblastoma involving the mandible in a 70-year-old patient. The case was managed by segmental mandibulectomy and flap repair. Unicystic ameloblastoma accounts for only 13% of all known cases in scientific literature. Considering the rarity of the lesion, the purpose of presenting this report on a clinical case is to emphasize the importance of radiological evaluation and histopathological examination for the diagnosis of ameloblastoma.Keywordsameloblastoma; odontogenic cysts; odontogenic tumors; segmental mandibulectomy.

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