Visual acuity was 20/20 OD and 20/400 OS, with central scotoma and pain in the left eye. There was vitritis with preretinal exudation and surrounding areas of focal retinitis and retinal hemorrhages (Figure 1A). Vitreous aspiration and intravitreal administration of amphotericin B (5 μg/0.05 mL) were performed in conjunction with intravenous voriconazole treatment (6 mg/kg). Four days later, 20 colonies of S apiospermum were identified, sensitive only to voriconazole (minimum inhibitory concentration [MIC], 0.5 μg/mL) and resistant to amphotericin B (MIC, >16 μg/mL), flucytosine (MIC, >64 μg/mL), and itraconazole (MIC, 2 μg/mL). The patient received 2 subsequent intravitreal injections of voriconazole (100 μg/0.1 mL), a vitrectomy with intravitreal voriconazole (150 μg), and 3 weekly intravitreal injections of voriconazole (100 μg). Cutaneous blood and lung cultures yielded S apiospermum, and voriconazole monotherapy was continued (4 mg/kg intravenously twice daily in the hospital and then 200 mg orally twice daily at home). Initial and repeated vitreous voriconazole levels (Fungus Testing Laboratory, University of Texas, San Antonio) were evaluated by high-performance liquid chromatography and exceeded the MIC (Table). The patient's visual acuity progressed to no light perception with eye pain, and she chose to undergo enucleation. Only the initial ocular culture had positive findings; results of all subsequent cultures were negative.