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We sought to illustrate the difficulty in managing uncommon, pigmented mold-related corneal ulceration and to highlight the role of itraconazole in treating these patients.We describe the management and clinical course of a patient with a recurring corneal infection caused by Fonsecaea pedrosoi and discuss this experience in the light of existing literature on management of cutaneous chromoblastomycosis.A corneal ulcer caused by this organism healed initially on treatment with topical and systemic antifungal medication, but infection recurred in the deep stroma 4 months after cessation of therapy. After failure to respond to a further period of medical therapy, a small therapeutic penetrating keratoplasty was performed. Culture of a fibrinous membrane from the anterior iris surface demonstrated intraocular fungal infection, and postoperatively, an episode of marked fibrinous uveitis developed, suggesting the presence of viable intraocular fungal elements. A large penetrating keratoplasty was therefore performed with excision of involved iris in combination with extracapsular cataract extraction. F. pedrosoi was again cultured from the fibrinous membrane adherent to the iris and from the anterior lens capsule. Postoperatively the patient received a 5-month course of systemic itraconazole, and no further recurrences have been encountered after a further 2 months.F. pedrosoi is the organism most commonly isolated from the chronic cutaneous mycosis, chromoblastomycosis, and is relatively resistant to medical therapy. As has been reported for cutaneous disease, surgery in combination with systemic itraconazole may provide the best chance of cure in corneal chromoblastomycosis.