Ameloblastic carcinoma (AC) is a rare and aggressive malignant epithelial odontogenic tumor that may arise either from the mandible (66.6%) or the maxilla (33.3%). Besides being formed de novo, the tumor can develop from a pre-existing ameloblastoma or odontogenic cyst. The clinical course of AC is typically aggressive with extensive local destruction. AC has also been reported to spread to various parts of the body through lymphatic system. Skull base location and intracranial extension of this tumor are uncommon. In this case report we present the clinical course, management and follow up of an uncommonly large AC that destructed the anterior skull base and extended into the intracranial space.

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