Mycotic keratitis usually occurs in conjunction with trauma to the cornea. Scedosporium apiospermum, a dematiaceous fungus linked to the teleomorph Pseudallescheria boydii is not a common agent of mycotic keratitis. A 22-year old male patient with mycotic keratitis due to S. apiospermum is presented. In in vitro susceptibility testing, the isolate showed resistance against amphotericin B (minimum inhibitory concentration [MIC] 16 microg ml(-1)) but was susceptible to itraconazole (ITC) and fluconazole with MICs of 0.125 microg ml(-1) and 4 microg ml(-1), respectively. The patient was cured clinically after ITC treatment and surgical intervention. Azoles may be superior for eliminating S. apiospermum from infected ocular sites.