The Scedosporium genus consists of filamentous fungi that inhabit soil, sewage, manure, and polluted waters, and contains two medically important species: Scedosporium apiospermum and Scedosporium prolificans. Scedosporiosis is caused by inhalation or traumatic subcutaneous implantation of the organism, and may have varied clinical presentations. A variety of pulmonary manifestations can occur, but to our knowledge, pneumothorax as initial presentation has not been reported. Treatment may be difficult, as the disease occurs more commonly in immunocompromised patients, and the organism may demonstrate resistance to various antifungal agents. We describe successful treatment of extensive pulmonary disease due to S apiospermum in a patient with acquired immune deficiency syndrome (AIDS).