OBJECTIVE To report on a human immunodeficiency virus (HIV)-infected patient with uveitis and an intraocular HIV1 RNA load largely exceeding that of plasma and no evidence of other intraocular infectious agents causing uveitis than HIV itself. METHODS Interventional case report. METHODS A 37-year-old male HIV-infected patient with uveitis and no retinal manifestations. METHODS Clinical and laboratory examinations including extensive intraocular fluid analyses for various pathogens and HIV-1 RNA loads in the aqueous and plasma. METHODS Results of aqueous analysis and ophthalmologic features. Correlations between the results of aqueous testing and clinical characteristics. RESULTS A 37-year-old patient presented with progressive uveitis. He was positive for HIV-1, and his HIV-1 RNA plasma load was 44,600 copies/mL. His intraocular HIV-1 RNA load was >1,900,000 copies/mL, which largely exceeded his concurrent plasma load. No evidence of infectious agents other than HIV itself was found, and the uveitis reacted promptly to solely the antiretroviral treatment. CONCLUSIONS Our findings suggest that HIV can locally replicate within the eye and cause an intraocular inflammatory reaction.