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Keratocystic odontogenic tumor (KCOT) is a benign odontogenic tumor with an aggressive behavior and high recurrence rate. The most common site of predilection is the posterior mandible. In contrast, KCOTs occurring in the maxillary region are relatively rare. However, the maxillary involvement poses a greater and increased threat, due to proximity to vital structures such as maxillary sinus, orbital floor, and infratemporal fossa. This report presents such a case of KCOT involving the maxillary sinus eroding the floor of the orbit and provides an account of the factors that need to be considered during management.

Recurrent keratocystic odontogenic tumor of right maxillary sinus involving the right infraorbital rim