We describe 2 patients with hematologic malignancy who developed endophthalmitis due to Trichosporon beigelii during the course of treatment with multiagent chemotherapy. Blood cultures revealed T beigelii for both patients. Although one of the patients was treated with fluconazole (FLCZ) and 5-fluorocytosine, the trichosporonous endophthalmitis was resistant to both drugs. This patient subsequently received amphotericin B (AMPH-B) therapy, and the eyes were treated with vitrectomy. The second patient also received AMPH-B for FLCZ-resistant trichosporonous chorioretinitis. In both patients, systemic treatment with AMPH-B successfully resolved the trichosporonous endophthalmitis that was resistant to multiple antifungal drugs. Endophthalmitis due to trichosporonosis is difficult to treat. The administration of AMPH-B is likely to be more effective in treating endophthalmitis due to trichosporonosis when the disease is at an early stage.