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Scedosporium prolificans is a saprophytic fungus responsible for an increasing number of infections among immunocompromised hosts. Most disseminated S. prolificans infections prove fatal due to persistent neutropenia, and inherited resistance to currently available antifungal drugs. The authors report a fatal case of a paediatric Korean patient who progressed to severe sepsis from S. prolificans infection after induction chemotherapy for acute lymphoblastic leukaemia. Treatment with itraconazole was unsuccessful and the patient died within 6 days of admission.