Background: Ameloblastoma is a rare benign odontogenic tumor with a metastasis rate estimated at 2% of cases, mainly involving the lung (80%) and lymph nodes (20%).
Methods: We hereby present the case of a 26 year old patient with a history of locally recurrent mandibular ameloblastoma who developed a temporal intracranial ameloblastoma tumor requiring a collaborative neurosurgical and maxillo-facial radical surgical approach.
Conclusion: Although ameloblastomas are histologically benign, the temporal topography questions the dissemination pathophysiology of the tumor (metastasis or local extension through temporal muscle fibers), mainly relevant in cases of multiple recurrences.
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