Keratocystic odontogenic tumor (KCOT) is a benign odontogenic neoplasm which is characterized by aggressive behavior and a high recurrence rate. KCOTs have a predilection for the angle and ascending ramus of the mandible. Maxillary KCOTs are usually associated with nevoid basal cell carcinoma syndrome. Very few cases of non-syndromic KCOT crossing maxillary midline have been reported. In this article, we report a rare case of KCOT which presented as a multilocular radiolucency crossing midline

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... nd involving periapical area of maxillary incisors. The lesion thus simulated a periapical cyst. However, the lesion was proved to be a KCOT on histopathology. Thus this report highlights the need for histopathological evaluation of periapical lesions for accurate diagnosis and treatment plan.