To report a patient with severe Chrysosporium keratitis successfully treated by voriconazole.Case report.A 37-year-old healthy male presented with irritation, pain and reduced vision in his left eye after mud contamination. Examination demonstrated corneal stromal infiltration, endothelial plaque and hypopyon. Corneal scrapings demonstrated numerous septate hyphae, and specimen cultures were positive for Chrysosporium sp. The lesion did not respond to aggressive topical 5% natamycin, 0.15% topical amphotericin B and oral itraconazole. The patient was then treated by topical 1% voriconazole every hour. Intracameral and intrastromal voriconazole injections (50 μg/0.1 mL) were also undertaken. The keratitis was significantly improved after voriconazole.To the best of the authors' knowledge, this is the first report on the use of voriconazole for Chrysosporium keratitis. Voriconazole may be an effective alternative to conventional antifungal agents in some cases of fungal keratitis. It should be considered before shifting to therapeutic keratoplasty.