Ameloblastic carcinoma is a rare odontogenic tumor which arises in the mandible or maxilla. It is classified into the primary and secondary types, and the latter one is extremely rare. We report herein on a case of secondary ameloblastic carcinoma in a 36-year-old man who complained of indolent swelling at the left side of the mandible. CT and MRI images revealed a multiocular mass with bone destruction in the mandible, and the existence of the ameloblastic carcinoma was confirmed with multiple biopsies. The lesion was resected en bloc with a 1.5 cm safety margin, and reconstructed with an osteocutaneous fibula flap. The histological features of the tumor were consistent with secondary ameloblastic carcinoma. Neither recurrence nor metastasis has been found for one year after the surgery. The clinical, radiological and histological features of the ameloblastic carcinoma are discussed with reference to the relevant literature.
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