Tehran University of Medical Sciences Journal of Craniomaxillofacial Research 2345-5489 9 3 2023 04 04 153 157 Ayoub Bakhil Department of Plastic and Maxillofacial Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco. Nawfal El Hafdi Department of Plastic and Maxillofacial Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco. Yasmina Ribag Department of Plastic and Maxillofacial Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco. Hicham Sabani Department of Plastic and Maxillofacial Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco. Lahcen Khalfi Department of Plastic and Maxillofacial Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco. Jalal Hamama Department of Plastic and Maxillofacial Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco. Karim El Khatib Department of Plastic and Maxillofacial Surgery, Mohammed V Military Hospital, Mohammed V University, Rabat, Morocco. 2022 11 24 2023 01 07 Introduction: Ameloblastic fibroma is a rare mixed odontogenic tumor, affects the young population, its management is mainly surgical. We report in this work the first observation of a concomitant bimaxillary localization. Materials and Methods: This is a 31-year-old female patient with no pathological history who presented to our department for management of a maxillomandibular tumor. The clinical examination revealed a poor oral condition and a swelling of the alveolar ridges. The CT scan of the facial mass revealed a multilocular cystic lesion encompassing teeth in the maxillary and mandibular bone. The biopsy came back in favor of an odontoameloblastic fibroma. Management consisted of radical resection with reconstruction using local flaps. FOA is a tumor distinct from ameloblastoma, it affects the young patients without any predilection to gender. The radiological image is a mono or multilocular cystic image which poses a problem of differential diagnosis with other cystic tumors. The management is surgical, clinical and radiological postoperative surveillance is primordial given the risk of recurrence or sarcomatous transformation. Conclusion: The FOA was for a long time considered as a form of ameloblastoma, is a rare tumor in the mandibular localization is the most frequent, the bimaxillary localization has never been described and the case we presented is the first in literature. Keywords: Ameloblastic fibroma; Maxillomandibular; Radical surgery; Surveillance. https://jcr.tums.ac.ir/index.php/jcr/article/view/423 https://jcr.tums.ac.ir/index.php/jcr/article/download/423/362