A rare case of plexiform ameloblastoma of right maxillary sinus

Abstract

Plexiform ameloblastoma is a benign odontogenic neoplasm characterized by locally invasive growth and high recurrence rate with incomplete resection. Plexiform ameloblastomas in the sinonasal tract, particularly the maxillary sinus, are exceptionally rare, comprising about 0.11% of sinonasal tumors, with a male predilection and later presentation age (mean 59.7 years) compared to jaw variants. A 40-year-old male presented with recurrent epistaxis and on examination revealed a reddish nasal mass. Initial CT suggested a right maxillary mucocele or low-grade neoplasm. FESS with total excision of the friable mass was done. Histopathology reported plexiform ameloblastoma. At 2-year follow-up, MRI revealed a recurrent 3.9×3.2×3.6 cm lesion with bony erosion, for which right infrastructural maxillectomy was done followed by obturator placement. The patient remains disease-free on subsequent follow-up. While conservative FESS may suffice initially, complete resection via maxillectomy is crucial to prevent early recurrence (typically 1-2 years post-surgery). Complete surgical excision offers excellent prognosis for sinonasal Plexiform Ameloblastoma, with long-term follow up necessary due to local aggressiveness. This case underscores the value of histopathology and serial imaging in management.