Ameloblastoma is an odontogenic tumor characterized by various sites of metastasis, malignant transformation, and a high recurrence rate over time. Ameloblastic carcinoma(AC) is the term reserved for an ameloblastoma with histologic evidence of malignancy in the primary tumor. AC is classified into two types: most ACs occur de novo, and only few cases of malignant transformation of ameloblastoma become apparent. Here, we report a case of AC, arising from recurrent acanthomatous ameloblastoma on the maxillary sinus, in a 60-year-old male patient. The mass was first diagnosed as acanthomatous ameloblastoma; subsequently, surgical curettage was performed thrice while partial maxillectomy was performed twice. On the fifth recurrence, the tumor was identified as AC.