Introduction: We report a case of posttraumatic keratomycosis caused by Scedosporium apiospermum that was treated with oral and topical voriconazole and penetrating keratoplasty. Case report: 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 mu g/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. Conclusion: 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.