Ameloblastic carcinoma is a rare primary tumor of the maxillofacial skeleton with a distinct predilection for the mandible. These lesions may initially show histologic features of ameloblastoma that dedifferentiate over time. Other ameloblastic carcinomas initially present with morphologic features suggestive of ameloblastoma with areas of epithelial dedifferentiation. We herein report a rare case of aggressive ameloblastic carcinoma in a 22-year-old white man who developed widespread bony metastases and expired 4 years after initial diagnosis.
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