Ameloblastoma is an epithelial odontogenic tumor with a benign nature and demonstrates local aggressiveness. It frequently occurs between the third and fifth decades of life, showing significant gender predilection. While typically displaying a benign growth pattern, it tends to invade and sporadically metastasize locally. Ameloblastoma is predominantly found in the posterior regions. Periodic recur commonly follows insufficient treatment. Hence, conducting thorough identification of tumors and management is crucial to prevent relapse. Complications and improved prognosis are associated with meticulous surgical techniques, regular follow-up care, and early detection of recurrence. This study presented a report of a 19-year-old male with swelling in the left lower jaw, detailing its area of complaint, radiographic findings, histopathologic characteristics, and different treatment approaches. The uniqueness of the case is the hybrid histopathology of ameloblastoma composed of plexiform and desmoplastic variants.

Hybrid ameloblastoma has a variable clinical, radiological, and histopathological presentation. They contain two or more different histologic types and their biologic comportment is still arguable. We herein present a case of a hybrid variant of desmoplastic ameloblastoma which is the first of its kind to have ever been reported due to its unusual location in the maxillary anterior region, along with systematic review of clinicopathologic features of reported cases immunohistochemical markers may act as an adjunct in the accurate diagnosis of these lesions.