PURPOSE: To describe a case of microsporidial keratoconjunctivitis in a patient without human immunodeficiency virus (HIV) infection. METHODS: Case report. An epithelial corneal scraping from a woman with chronic bilateral keratoconjunctivitis was evaluated by Giemsa stain. RESULTS: Giemsa stain of an epithelial corneal scraping disclosed intracellular and extracellular spores characteristic of microsporidia. An HIV enzyme-linked immunosorbent assay (ELISA) test was negative. The signs and symptoms of the bilateral keratoconjunctivitis resolved after treatment with albendazole. CONCLUSION: Microsporidia may cause a chronic epithelial keratoconjunctivitis in the absence of HIV infection.