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Ameloblastic carcinoma (AC) is an exceedingly rare odontogenic cancer about which there is limited information in the literature. We present a case of AC originating in the sinus cavity and extending to the skull base in a patient in the first trimester of pregnancy. Diagnostic work up was complicated by this pregnancy, which delayed radiation exposure with imaging. Once scans were obtained, diagnosis was further complicated by the radiographic similarities between possible lung metastases and previously undiagnosed sarcoid nodules. After thorough work up to rule out metastatic disease, the patient was successfully treated with primary surgical resection followed by adjuvant chemoradiation. The patient remained disease free at one year after therapy. This case demonstrates the importance of thorough work up in the diagnosis of AC, and is an opportunity to review the literature and discuss therapeutic methods to treat this rare, aggressive neoplasm.

A Rare Presentation of Ameloblastic Carcinoma of the Sinus Cavity and Skull Base