Intraocular infection due to Blastomyces dermatitidis is rare, and only 10 cases have previously been reported. Manifestations of ocular blastomycosis can range from keratitis to panophthalmitis, and it is often difficult to diagnose ocular blastomycosis early. We report the case of a 45-year-old man who had disseminated blastomycosis that involved the lungs, skin, and ocular uvea and who was successfully treated with systemic and local antifungal therapy. We also review the literature describing the spectrum of clinical findings due to intraocular blastomycosis. © 1994 by The University of Chicago.