Scedosporium infections are rare complications in immunocompromised patients or patients with chronic pulmonary disease. While Scedosporium prolificans is resistant to most antimycotics, Scedosporium apiospermum is usually sensitive to voriconazole and posaconazole. Pharmacokinetics and efficacyof nebulized voriconazolehavebeendescribedina murinemodelpreviously. We report for the first timethesafeandeffectiveuse of nebulized voriconazole for the treatment of severe pulmonary infection with Scedosporium apiospermum in an adolescent with cystic fribrosis.