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A 79-year-old male presented with recurrent maxillary ameloblastoma with intracranial invasion into the left orbit, previously histologically diagnosed as benign ameloblastoma. Skull x-ray films and computed tomography showed the multicystic mass had destroyed the skull base. The tumor was nearly completely removed. However, microscopic examination revealed residual tumor cells around the left optic nerve. Histological examination found no malignant transformation in the tumor specimen. Aggressive complete removal of maxillary ameloblastoma should be attempted even in cases of intracranial invasion.
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