We report a case of posttraumatic keratomycosis caused by Scedosporium apiospermum that was treated with oral and topical voriconazole and penetrating keratoplasty.A patient was admitted to the hospital with a corneal abscess of his right eye due to trauma while gardening. No improvement was noted with topical fortified antibiotics (vancomycin, gentamicin, and cefazolin) and antimycotic (amphotericin B 1%) and oral itraconazole (200 mg/day). Fungal cultures of corneal scrapings revealed growth of Scedosporium apiospermum, a strain for which the main antifungals showed high minimal inhibitory concentrations (MICs), whereas the MIC of voriconazole was 0.125 microg/mL. Despite some improvement with topical 1% voriconazole and oral voriconazole (200 mg/day) treatment, a therapeutic penetrating keratoplasty was performed because of the high risk of corneal perforation. The graft remained clear without fungal recurrence with topical 2% cyclosporine A, dexamethasone, and voriconazole treatment.Scedosporium apiospermum is an uncommon cause of mycotic keratitis in humans. Prognosis is generally poor because of delayed diagnosis and resistance to conventional antifungals. Voriconazole is a triazole broad-spectrum antifungal agent. In conjunction with its oral administration, topical application of voriconazole extends the current armamentarium of antifungal agents for keratomycosis.