Background. Brucellosis has unusual clinical manifestations. Ocular involvement caused by brucellosis remains poorly recognized in areas in which brucellosis is endemic. Methods. A prospective study was performed to evaluate patients attending the Instituto de Medicina Tropical "Alexander von Humboldt" and the Hospital Nacional Cayetano Heredia (Lima, Peru) from January 1980 through December 2005 who received a diagnosis of brucellosis with ocular involvement. Diagnosis was made on the basis of clinical findings as well as agglutinations and/or culture positive for Brucella melitensis. Results. During a period of 26 years, 1551 patients with brucellosis were seen, including 52 patients with ocular brucellosis. We found that 7 (0.7%) of 965 patients with acute brucellosis and 45 (7.9%) of 570 patients with chronic brucellosis had ocular brucellosis (P < .001). In 16 patients with brucellosis, the disease stage was unclassified. The most frequent ocular presentation was uveitis, which was found in 43 (82.7%) of the 52 patients with ocular brucellosis. Posterior uveitis was the most frequent uveal syndrome (21 cases; 45.7%). Patients with panuveitis had the worst visual prognosis: 8 of 9 patients with panuveitis were legally blind, including 5 patients with no light perception. Conclusions. Brucellosis may involve the eye and can lead to serious complications. In patients with brucellosis, early ophthalmologic evaluation can lead to prompt treatment and might prevent blindness from severe ocular damage. © 2008 by the Infectious Diseases Society of America. All rights reserved.