The fungus Scedosporium prolificans was first described as a human pathogen in 1984, and has been associated with metastatic endophthalmitis and one previously reported case of sclerokeratitis. We report a case of S. prolificans sclerokeratitis in the setting of late scleral necrosis complicating pterygium surgery with adjuncive beta-irradiation. A poor clinical response to topical natamycin and amphotericin B, and systemic itraconazole and ketoconazole was encountered. Enucleation was required, with subsequent microbiological cure. Pathological correlation is described. S. prolificans infections often respond poorly to medical therapy. Early surgical intervention is indicated in culture-proven scleritis due to Scedosporium prolificans