Uveitis is a well-known late complication of systemic leptospirosis, a zoonotic disease caused by the water-borne spirocheteLeptospira.Although it is one of the world's most widespread febrile diseases, it remains underdiagnosed, mainly because of protean manifestations, lack of awareness, and nonavailability of laboratory support. Systematic collection of published literature was conducted using Medline, the Cochrane library, and bibliographies of retrieved reports. Articles directly applicable to ocular leptospirosis and current reports on the epidemiology, basic research, clinical presentations, and management of leptospirosis were reviewed. Changing trends in risk factors and an expanding spectrum of ocular and systemic findings have been reported. Molecular research on leptospirosis has shown remarkable progress; several rapid diagnostic modalities are currently under study. Awareness of this entity is absolutely essential to arrive at an accurate diagnosis and to prevent its potential reversible and irreversible ocular complications.