Abstract
BACKGROUND: Ameloblastic carcinoma (AC) represents a rare and aggressive malignant odontogenic tumor that derives from the epithelial tissue of the jawbone, predominantly affecting the mandible. This report describes a case of ameloblastic carcinoma that occurred after removal of a right-sided maxillary ameloblastoma. CASE PRESENTATION: A 17-year-old male teenager visited author's affiliation with a complaint of a two-week history of spontaneous pain around upper right premolar area after a primary ameloblastoma resection. Intraoral examination revealed that an ulceroproliferative growth with everted margins was noted over the retromolar region adjacent to #17. An occupying lesion was observed in the right side of maxilla by maxillofacial imaging. The patient underwent partial maxillectomy, elective level I-II neck dissection, and immediate reconstruction with submental island flap. Histopathological assessment post-right maxillectomy identified a high-grade AC with malignant spindle cell transformation. Post-treatment follow-up for 6 months showed no recurrence of the malignancy. CONCLUSION: Primary ameloblastoma or AC is increasingly being reported, but secondary AC of the maxilla is rarely described in detail. Although the underlying causes of this condition are still not well understood, intensive treatment and careful follow-up are key steps in preventing the disease from recurrence and malignant changes.