Pediatric diamond-blackfan anemia after hematopoietic stem cell transplantation complicated by bronchiolitis obliterans and air-leak syndrome leading to lung transplantation: a case report with multimodal follow-up
Introduction: Bronchiolitis obliterans syndrome (BOS) is a severe, often fatal pulmonary manifestation of chronic graft-versus-host disease (cGVHD) following allogeneic hematopoietic stem cell transplantation (HSCT). Its progression to air-leak syndrome (ALS) signifies a critical deterioration with exceedingly high mortality. Lung transplantation (LTx) remains a rare salvage option, especially in children, with scarce reports of successful outcomes in those with this complication cascade. Case presentation: /FVC 52%), and high-resolution computed tomography (HRCT) revealed mosaic perfusion and bronchial wall thickening, contributing to the diagnosis of BOS. Despite intensive immunosuppressive therapy, she developed ALS on +360d and type II respiratory failure on +475d. Sequential bilateral LTx was performed on October 28, 2025. Postoperatively, the patient recovered following the management of multidrug-resistant bacterial infections and respiratory complications, with no rejection or recurrence of cGVHD during follow-up. Conclusion: This report presents the youngest documented DBA case of successful LTx for BOS complicated by ALS after allo-HSCT globally. It demonstrates that dynamic multimodal monitoring is crucial for early BOS detection. LTx is a viable therapy for end-stage pulmonary cGVHD in children. This case underscores the need for proactive monitoring in high-risk patients and provides a paradigm for managing this complex complication.
Authors
- Hairong Fei
- Shu Wang (ORCID: https://orcid.org/0000-0001-5447-0946)
- Yanxia Zhao (ORCID: https://orcid.org/0000-0003-0342-2578)
- Nan Ge (ORCID: https://orcid.org/0000-0003-1025-9756)
- Feng Hou (ORCID: https://orcid.org/0000-0002-1205-0131)
- Xue Shi (ORCID: https://orcid.org/0000-0001-6293-8991)
- Wei Wang
- Xuewei Li
- Chunting Zhao
- Xiaodan Liu
- Wenhui Zhang
Institutions
- Qingdao University (CN)
- Affiliated Hospital of Qingdao University (CN)
Publication Details
- Journal
- Frontiers in Immunology
- Published
- 2026-04-22
- DOI
- https://doi.org/10.3389/fimmu.2026.1782188
- Primary Topic
- Transplantation: Methods and Outcomes
- Type
- article
- Field-Weighted Citation Impact
- 0.00