Odontoameloblastoma (OA) is an extremely rare mixed odontogenic neoplasm characterized by amelobastomatous component and odontoma–like elements in the same tumor mass. It occurs predominantly in chidren and young adults. The majority of cases are associated with unerrupted teeth. Till date very few cases of Odontoameloblastoma have been reported in the literature on base the criteria of the current World Health Organization classification of odontogenic tumors. According to the cases reported in the English literature ^[9]; among26 cases, only 5 cases have been reported involving posterior maxillary region. In this article we present extremely rare case of odontoameloblastoma in 8 year old female patient with mild swelling and facial deformity in left maxillary jaw region.