We report the first case of pulmonary scedosporiosis detected by next-generation sequencing (NGS) from bronchoalveolar lavage fluid (BALF) in a 67-year-old male with bronchiectasis and hematopoietic failure. Scedosporium apiospermum is a ubiquitous organism present in the environment with intrinsic resistance to many antifungal agents. The patient developed respiratory failure, pulmonary consolidation, and septic shock shortly thereafter, and responded poorly to antifungal therapy. This case highlights the combined application of NGS and traditional fungal culture in the clinical diagnosis of pulmonary invasive fungal disease. NGS is proposed as an important adjunctive diagnostic approach for uncommon pathogens. Keywords: Scedosporium apiospermum, Pulmonary scedosporiosis, Next-generation sequencing