PURPOSE: To report a case of microsporidial keratoconjunctivitis in an HIV-seronegative. METHODS: We report on a 40,year-old woman who presented with acute symptoms of 4 days' duration. Visual acuity was 6/6 in the right eye and 6/6 partial in the left eye. In the left eye, there was diffuse conjunctival congestion with papillary changes. There were multiple raised epithelial lesions in the cornea. RESULTS: Corneal scrapings stained with potassium hydroxide and calcofluor white showed Microsporidium spores in large numbers. These spores with polar staining were also confirmed in Gram stain. One month after initiation of itraconazole, the left eye was quiet. She had a visual acuity of 6/6 in that eye, with faint corneal haze. CONCLUSIONS: We report a rare case of microsporidial keratoconjunctivitis in a HIV-seronegative patient who was treated by debridement and oral itraconazole. Microsporidium should be suspected even in an immuno. competent individual if the corneal examination revealed showed multiple raised epithelial lesions. (C) 2003 by Elsevier Inc. All rights reserved.