Keratocystic odontogenic tumor is derived from the proliferation of residues of the dental lamina. Keratocystic odontogenic tumors have high recurrence rate from 0% to 62%, depending on the locations and types of treatment. The controversy still exists about treatment methods ranging from simple curettage to highly invasive en bloc resection. Furthermore, there is no consensus on the most effective surgical technique. We report the first case of removal via endonasal endoscopic approach for a huge, expansile keratocystic odontogenic tumor in the maxillary sinus extending to contralateral central incisor.
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