A 60-year-old white man from an area endemic for Histoplasma capsulatum presented with a vitreous wick, hypopyon iritis, and dense vitreitis 2 months after removal of an anterior chamber intraocular lens (IOL) for chronic iritis. A diagnostic vitrectomy was performed and H. capsulatum was cultured and identified 2 weeks later. Despite intravitreal and intravenous amphotericin as well as repeat vitrectomies, the inflammation worsened and the eye was removed. Results of histopathologic examination showed Histoplasma organisms along the vitreous wick, over the surface of the iris and ciliary body, and over the retina. No organisms were found in the choroid. Dalen-Fuchs-type nodules similar to those of sarcoid also were noted, but there was no evidence of granulomatous inflammation in the uvea. Because of his unilateral disease with Histoplasma in the vitreous wick, negative serology, and an absence of systemic infection, the authors believe that this patient had a previously unreported form of ocular Histoplasma, exogenous postoperative Histoplasma endophthalmitis.