Fungi are among the most common agents of microbial keratitis, especially in developing countries and tropical regions. Early diagnosis and appropriate antifungal treatment are critical for clinical success in fungal keratitis (FK). A 35-year-old female patient was referred to our hospital because of the diagnosis of keratitis with resistant to topical antibacterial treatment. Her visual acuity was counting fingers at one meter for the right eye. Biomicroscopic examination revealed a large, white keratitis center with peripherally extension on cornea. Fungal hyphae were observed in microscopic examination and a filamentous fungus was isolated in culture of the corneal scrapings. It was identified as Scedosporium apiospermum by both phenotypic and molecular methods. She was treated with topical and oral voriconazole according to susceptibility testing result. It is important to early diagnosis, determine the causative agent and perform a susceptibility testing in FK.Keywords: Keratitis, Scedosporium apiospermum, voriconazole.