| Summary: | Adenomatoid odontogenic tumor (AOT) is an uncommon nonaggressive tumor of the odontogenic epithelium with duct-like structures and varying degrees of inductive changes in the stroma. It comprises only 0.1% of tumors of the jaw and 3% of all odontogenic tumors. The majority of the cases (88%) are diagnosed in the second and third decades of life. The incidence is higher in males than in females. The tumor has a predilection for the anterior maxilla, most commonly associated with maxillary canine; however, involvement of the maxillary antrum is extremely rare. We report an unusually large AOT of the maxilla along with the computed tomography images in a 17-year-old female patient encroaching the maxillary sinus, orbital floor, nasal septum, ethmoidal sinus on the right side. It was also associated with an impacted canine. The patient was subjected for complete enucleation along with the removal of the impacted tooth. To the best of our knowledge, it is the first time that such a large AOT with involvement of the ethmoidal sinus has been reported.
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