The keratocystic odontogenic tumor, although a benign lesion, is peculiarly aggressive with a high recurrence rate. Its involvement with the maxillary antrum is atypical. We report the unusual case of a 20-year-old male patient with an extensive antral tumor associated with an impacted third molar, which was initially misdiagnosed as a dentigerous cyst. Clinical, radiographic, and histopathologic aspects were analyzed to provide useful information for the correct diagnosis, treatment, and prognosis within a multidisciplinary approach.