Background: Rifabutin-associated uveitis has been recognized and established overseas, but there have been no reports of its occurance in Japan. We report a case of rifabutin-induced hypopyon uveitis in a patient with acquired immunodeficiency syndrome (AIDS).

Case: A 45-year-old Filipino man with AIDS and pulmonary tuberculosis was on highly active antiretroviral therapy (HAART; lopinavir/ritonavir, zidovudine/lamivudine) and anti-tuberculosis therapy (isoniazid, rifabutin). At 11 weeks after starting rifabutin administration, unilateral acute uveitis with hypopyon developed, which progressed to bilateral uveitis by the following day. Funduscopic examination revealed no vitreo-retinal abnormalities. The uveitis resolved after discontinuation of the rifabutin and initiating topical corticosteroids and mydriatic therapy. No recurrence of the uveitis was noted during a 5-month follow-up period.

Conclusion: Rifabutin-associated uveitis with hypopyon should be considered as one possible causes of uveitis.

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