We herein discuss the clinical course of a keratomycosis case with the rarely encountered etiology of Scedosporium apiospermum. A 22-year-old male was referred with the diagnosis of unhealed corneal ulcer, foreign body sensation, pain, and photophobia and decreased vision in his left eye. After visualization of hyphal elements, antifungal therapy containing amphotericin B was given promptly both parenterally (1.5 mg/kg/day, intravenously) and topically (1% solution). Amphotericin B was discontinued and itraconazole (400 mg/day bid) was started because of preliminary identification of the isolate that it might be a Scedoporium apiospermum. Unfortunately, on the fifth day of itroconazole therapy, almost the entire cornea was totally melted and almost disappeared. The patient underwent penetrating keratoplasty, lens extraction and intraocular lens implantation surgery with triple procedure on the 10th day of itraconazole therapy. The donor cornea was clear and there was no inflammatory sign on the eye, and the patient was discharged with a vision of 0.6 according to snellen acuity chart. However a fibrovascular membrane developed later and filled anterior chamber of the patient's left eye. The patient underwent a second wide (limbus to limbus) penetrating keratoplasty after the excision of the fibrovascular membrane, and the development of corneal edema. Retinal detachment developed after the development of second fibrovascular membrane and donor corneal insufficiency, and the eye was left to phitisis. Although Scedoporium apiospermum is a rare reason for keratomycosis, it must be taken into account in the differential diagnosis. Surgeon must keep in mind that when wide penetrating keratoplasy is needed for total corneal melting, recurrent fibro-vascular membrane could develop due to limbal insufficiency. New treatment options are needed to prevent the development of fibrovascular membranes.

Keywords: Fungal keratitis, triple penetrating keratoplasty, neovascular membrane, limbal insufficiency, hiperbaric oxygen therapy