Beyond the Midline: An Unusual Presentation of Acanthomatous Ameloblastoma in the Anterior Maxilla

Acanthomatous ameloblastoma is an uncommon histopathological variant of ameloblastoma characterised by extensive squamous metaplasia and keratin formation within odontogenic epithelial islands.It predominantly affects adults between 30 and 60 years of age and most commonly occurs in the posterior mandible; involvement of the anterior maxilla is rare.A 55-year-old man presented with a progressively enlarging swelling in the anterior maxilla accompanied by pain during mastication.Intraoral examination revealed a diffuse swelling that crossed the midline, involved the labial vestibule, and caused expansion of the labial cortical plate.Cone-beam computed tomography demonstrated a multilocular lesion with cortical perforation, external root resorption, tooth displacement, and involvement of the nasopalatine canal.Histopathological examination confirmed acanthomatous ameloblastoma.The patient underwent partial maxillectomy with complete surgical excision.Postoperative healing was uneventful, and no clinical or radiographic evidence of recurrence was detected during two years of follow-up.This case highlights the importance of correlating clinical, radiographic, and histopathological findings for accurate diagnosis and appropriate management of maxillary ameloblastoma.Long-term surveillance remains essential because of its locally aggressive behaviour and potential for late recurrence.