To report a case of drug-resistant Fusarium deep keratitis following sclera graft.

A 48-year-old woman, who underwent excision of the pterygium nine years previous, received a scleral graft for scleromalacia and ocular pain. After surgery, due to melting of the scleral graft and the presence of Fusarium deep keratitis, she received surgical and intensive medical treatment including three intracameral injections of antifungal agent, administration of topical voriconazole 1%, scleral re-graft, autologous conjunctival graft, and amniotic membrane transplantation. Nevertheless, the patient's ocular condition deteriorated with increased hypopyon, corneal opacity, and perforation. Enucleation was ultimately performed.