PurposeThis study reports a case of bilateral rifabutin-associated uveitis in a child with a history of acute myeloid leukemia.MethodsWe utilized a clinical case description and brief discussion.ResultsA 17-year-old girl presented with acute bilateral anterior uveitis, a hypopyon in the left eye, and moderate bilateral vitritis. She had a history of acute myeloid leukemia status post-allogeneic hematopoietic stem cell transplant 5 years earlier. She was receiving rifabutin for a biopsy-proven Mycobacterium avium complex pulmonary infection. Work up for infectious and neoplastic etiologies was negative. The uveitis initially responded to topical corticosteroids, but recurred when the drops were tapered. Fluorescein angiography demonstrated diffuse vasculitis of small retinal vessels and cystoid macular edema. After rifabutin was discontinued, the uveitis and vasculitis slowly resolved.ConclusionFluorescein angiography demonstrated widespread retinal vasculitis which is a rare manifestation of rifabutin-associated uveitis.