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The purpose of this study was to determine the incidence and the high-resolution computed tomography findings of air-leak syndromes in a large group of hematopoietic stem cell (HSC) transplant recipients with proved chronic graft versus host disease (cGVHD).High-resolution computed tomography scans of 9 allogeneic HSC transplant recipients (8 men, 1 woman; 19 to 45 years of age; median 28 y), with a proven diagnosis of cGVHD-related bronchiolitis obliterans syndrome (BOS) were reviewed for the presence, appearance, and distribution of abnormalities.Nine patients with cGVHD developed 10 episodes of spontaneous air-leak syndromes secondary to cGVHD-related BOS during the study period analyzed; for a 2-year estimated cumulative incidence of 2.1% (95% confidence interval 0.2% to 3.8%) its prevalence was 5.7% (95%CI: 2.6% to 10.5%). All cases were allogeneic HSC transplant recipients with cGVHD who acquired new respiratory symptoms and/or radiologic abnormalities. Pneumomediastinum and pneumothorax were present in 6 patients. Subcutaneous emphysema was identified in 3 patients and pulmonary interstitial emphysema in 2 patients. A combination of different air-leak syndromes was observed in 6 patients.Air-leak syndromes represent an uncommon late complication in HSC transplant recipients with cGVHD-related BOS.