Summary:Oncocytic tumors of the lacrimal sac are very rare, with only 21 cases documented in the world literature. The authors report a 75-year-old woman who sought treatment for recurrent dacryocystitis and a palpable mass in the left medial canthal area. The patient underwent surgery for removal of a suspected lacrimal sac cancer, but pathologic findings indicated benign oncocytic adenomatous hyperplasia. Complete removal of the tumor and long-term follow-up are necessary in patients with lacrimal sac oncocytic lesions. The authors discuss the characteristic features of ocular adnexal oncocytic lesions and review the literature.