Tehran University of Medical Sciences Journal of Craniomaxillofacial Research 2345-5489 7 2 2020 10 03 98 101 Mahdi Azadi Department of Oral and Maxillofacial Surgery, School of Dentistry, Tehran University of Medical Sciences, Tehran, Iran. Farnoosh Mohammadi Department of Oral and Maxillofacial Surgery, School of Dentistry, Tehran University of Medical Sciences, Tehran, Iran. Narges Hajian Department of Oral and Maxillofacial Surgery, School of Dentistry, Tehran University of Medical Sciences, Tehran, Iran. 2020 07 14 Ameloblastoma is one of the most common types of oral odontogenic tumors. As per literature, ameloblastoma mostly occurs in the mandible butthe maxillary ameloblastoma has a more aggressive behavior due to anatomical features. Also, unicystic ameloblastoma may have lower recurrent rate. In this case report, we present a 60-year-old male patient with a history of unicystic ameloblastoma, which the intraluminal adenomatoid odontogenic tumor excisional biopsy surgery was performed but the patient didn’t follow the treatment completely, and after two years he came back with swelling of the right upper alveolar ridge. After the second surgery, the histopathologic report revealed a mixed plexiform-follicular ameloblastoma recurrence and it seemedthat previous surgery was not sufficient and more radical treatment is needed for the lesion. Keywords: Ameloblastoma; Maxilla; Adenomatoid odontogenic tumor. https://jcr.tums.ac.ir/index.php/jcr/article/view/334 https://jcr.tums.ac.ir/index.php/jcr/article/download/334/296