ABSTRACT: BACKGROUND: A case report to highlight the association between rifabutin and hypopyon METHODS: A 56 year old male presented with a one day history of blurred vision in his right eye. He had an established diagnosis of Crohn's disease which was in remission following treatment with rifabutin and clarithromycin. A brisk anterior uveitis with hypopyon and a mild vitritis was detected in the right eye. The acute inflammatory episode resolved following treatment with topical corticosteroids and withdrawal of rifabutin. RESULTS: The presence of hypopyon is atypical in uveitis associated with inflammatory bowel disease. The association between rifabutin treatment and hypopyon uveitis is well recognised in Mycobacterium avium paratuberculosis. However, use of rifabutin in the management of Crohn's disease is controversial and not widely known to an ophthalmic readership. CONCLUSION: This report highlights the importance of keeping abreast of novel therapeutic developments in systemic conditions likely to be encountered in ophthalmology.