Arash Sarrafzadeh
1 
, Parto Nasri
2 , Salar Nasr Esfahani
3 , Maryam Mohebiniya
4* , Soheila Jadidi
5* 1 Department of Oral and Maxillofacial Surgery, Shahid Beheshti University of Medical Sciences, Taleghani Hospital, Tehran, Iran
2 Department of Pathology, Isfahan University of Medical Sciences, Isfahan, Iran
3 Department of Pathology, School of Medicine, Arak University of Medical Sciences, Arak, Iran
4 Department of Oral and Maxillofacial Radiology, School of Dentistry, Arak University of Medical Sciences, Arak, Iran
5 Dental Research Center, Dentistry Research Institute, Tehran University of Medical Sciences, Tehran, Iran
Abstract
Ameloblastoma is the most common benign odontogenic epithelial tumor, accounting for approximately 11–18% of all odontogenic neoplasms and 1-3% of all oral cysts and tumors. It primarily affects the posterior mandible and usually arises in the third to fifth decades of life. Despite its slow growth, this tumor is locally aggressive and may metastasize or undergo malignant transformation. Histologically, ameloblastoma comprises several variants. The acanthomatous variant, although relatively rare, is characterized by squamous metaplasia and keratin formation within the central stellate reticulum-like cells of the tumor islands. The clinical and radiographic characteristics of ameloblastoma are often nonspecific and may resemble other multilocular radiolucent lesions. Accurate diagnosis necessitates the integration of advanced radiographic imaging, particularly cone-beam computed tomography, along with definitive histopathologic evaluation. We presented a rare case of acanthomatous ameloblastoma affecting both the anterior and posterior regions of the left mandible in a 33-year-old male with a five-year history of jaw expansion and progressive tooth mobility, along with its surgical management. The lesion showed extensive anterior extension, crossing the midline. This case underscores the significance of comprehensive differential diagnosis and histopathological confirmation in managing unusual presentations of ameloblastoma.