Purpose.To describe a case of fungal keratitis caused by a rare coelomycete pathogen,Colletotrichum gloeosporioides.Methods.An 82-year-old man with myelodysplastic syndrome developed fungal keratitis 6 months after cataract surgery in the left eye. Because the findings of the anterior chamber deteriorated after the initiation of natamycin, additional treatment with topical and systemic fluconazole was initiated.Results.The isolates of corneal scraping grewC. gloeosporioides. After antifungal treatment of 2 months, the corneal lesion resolved with no recurrence of infection over a 5-year follow-up period.Conclusions.A combination treatment of natamycin and fluconazole was effective in the treatment ofC. gloeosporioides.