OBJECTIVE To describe the first use of intravitreal voriconazole in a human eye for the treatment of Aspergillus endophthalmitis. METHODS Interventional case report. METHODS A 22-year-old woman receiving immunosuppressive agents 5 weeks after lung transplantation who presented with blurred vision and redness in the right eye. METHODS Intravitreal injection of voriconazole (100 microg/0.1 ml) with pars plana vitrectomy, given after isolation of A. terreus in the vitreous sample. Previous treatment modalities, including vitrectomy with repeated intravitreal amphotericin B and systemic voriconazole, failed to prevent deterioration. METHODS Visual acuity (VA) and ocular inflammation. RESULTS Significant improvement was observed in VA (to 6/15) and in ocular inflammatory reaction. The patient recovered with no evidence of systemic fungal infection. CONCLUSIONS Intravitreal voriconazole may be used as an adjunct to systemic treatment in patients with Aspergillus endophthalmitis. Further clinical studies are needed to determine how often this approach can safely treat this condition.