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 Ameloblastoma is an odontogenic tumor with diverse clinical behavior and histomorphologic presentations. Basal cell ameloblastoma are extremely rare variants of ameloblastoma. Keratocystic odontogenic tumor (KCOT) is a distinct form of odontogenic cyst that is considered a benign cystic neoplasm and not a cyst. Co-occurrence of these odontogenic tumors has been rarely reported.This paper reports a 34-year old female patient with a unilocular radiolucency around the crown of a partially erupted third molar that was detected accidentally on a routine radiographic examination. She underwent excisional biopsy with a clinical diagnosis of dentigerous cyst. Histopathologic examination revealed a cystic lesion with criteria of OKC and islands and nests reminiscent of basal cell ameloblastoma and acanthomatous ameloblastoma in the stroma. Follow-up showed no recurrence of lesion to date. Due to the rarity of the lesion, the diagnosis was challenging but altogether we made a diagnosis of keratoameloblastoma with basaloid features.

Keywords: ameloblastoma, odontogenic cyst, odontogenic tumor

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