We report here the first documented case of ocular coccidioidomycosis in a chimpanzee (Pan troglodytes). In 1996, a 12-year-old female chimpanzee was undergoing treatment with an experimental triazole, BayR3783, for coccidioidomycosis when she was diagnosed with severe conjunctivitis in the right eye. Subsequent development of a coccidioidal granuloma of the ventral conjunctiva and anterior uvea was noted over the next several months, distorting the lens, iris, pupil, and sclera and progressing to uveitis. Treatment with BayR3783 and subconjunctival injections of triamcinolone were successful in reducing the ocular mass, but extensive damage was done to the lens and cornea. This case study provides an interesting comparison to ocular coccidioidomycosis cases observed in both humans and canines.