We describe a case of keratitis caused by a new species of the hyphomycetous genus Sarcopodium, S. oculorum. The corneal ulcer developed after 5 months of treatment with corticosteroids in a Brazilian boy diagnosed with allergic conjunctivitis. Fungal hyphae and conidia were detected in corneal scrapings, and repeated cultures were positive for this fungus. The infection was resolved with natamycin and ketoconazole. Eleven antifungals were tested against this fungus, and all except flucytosine and fluconazole showed in vitro activity.