Carcinomas derived from ameloblastomas have been designated by a variety of terms, including malignant ameloblastoma, ameloblastic carcinoma, metastatic ameloblastoma, and primary intra-alveolar epidermoid carcinoma. The term of ameloblastic carcinoma is differentiated from the term of malignant amelblastoma and is defined as an ameloblastoma in which there is histologic evidence of malignancy in the primary tumor or the recurrent tumor(or metastasis), regardless of whether it has metastasized. The well-documented and adequately followed cases are currently lacking and this report described an instance of ameloblastic carcinoma with good result after treatment and review of literature.