The keratocystic odontogenic tumor (KCOT) is a frequently encountered developmental cyst of the jaws. The occurrence of KCOT in the maxillary sinus is rare. The mucosa of the maxillary sinus is susceptible to infections, allergic diseases, and neoplasm. The anatomic position of maxillary premolar and molar teeth is in close contact with the sinus predispose to spreading of pulp and periodontal infection, odontogenic cyst, and tumors to the sinus. Diagnosis and treating KCOT in maxillary sinus is challenging as treatment has to be rendered for sinusitis because of pathology in the sinus and for KCOT. We report a case of 35-year-old female with KCOT involving the lining of the maxillary sinus and put forward hypotheses for the origin of KCOT in the maxillary sinus.
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