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极迟发性慢性移植物抗宿主病,表现为闭塞性细支气管炎和胸膜实质纤维弹性组织增生症。

Extremely late-onset chronic graft-versus-host disease presenting with bronchiolitis obliterans and pleuroparenchymal fibroelastosis.

作者信息

Akiyama Sae, Murayama Chisa, Aizawa Saori, Mizushima Arei, Maeda Yukiko, Taniguchi Natsuko, Nagai Katsura, Harada Toshiyuki

机构信息

Center for Respiratory Diseases Japan Community Healthcare Organization Hokkaido Hospital Sapporo Japan.

出版信息

Respirol Case Rep. 2023 Jul 31;11(9):e01201. doi: 10.1002/rcr2.1201. eCollection 2023 Sep.

DOI:10.1002/rcr2.1201
PMID:37534048
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10390817/
Abstract

A 35-year-old woman experienced left back pain after a 2-h flight. She reported coughing and left back pain 1 day later when she presented to our hospital. Chest computed tomography showed pneumothorax of the left lung, bronchiectasis, thickening of the bronchial wall, nodules, and cavity lesions in both lungs. A pulmonary function test revealed obstructive ventilation disorder with normal lung diffusing capacity. She had a history of haematopoietic stem cell transplantation (HSCT) at 2 years and 3 months of age during the second disease remission of acute myeloid leukaemia. She was diagnosed with chronic graft-versus-host disease (cGVHD) presenting with bronchiolitis obliterans (BO) and pleuroparenchymal fibroelastosis (PPFE). To our knowledge, this is the first reported case of BO and PPFE diagnosed more than 30 years after HSCT.

摘要

一名35岁女性在乘坐2小时航班后出现左背部疼痛。1天后她前来我院就诊,自述咳嗽及左背部疼痛。胸部计算机断层扫描显示左肺气胸、支气管扩张、支气管壁增厚、双肺结节及空洞病变。肺功能测试显示为阻塞性通气障碍,肺弥散功能正常。她在急性髓系白血病第二次疾病缓解期,于2岁3个月时接受过造血干细胞移植(HSCT)。她被诊断为慢性移植物抗宿主病(cGVHD),表现为闭塞性细支气管炎(BO)和胸膜实质纤维弹性组织增生症(PPFE)。据我们所知,这是首例报道的HSCT后30多年诊断出BO和PPFE的病例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ce0/10390817/9cc7cf134f5e/RCR2-11-e01201-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ce0/10390817/43eda406a3f1/RCR2-11-e01201-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ce0/10390817/9cc7cf134f5e/RCR2-11-e01201-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ce0/10390817/43eda406a3f1/RCR2-11-e01201-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ce0/10390817/9cc7cf134f5e/RCR2-11-e01201-g003.jpg

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本文引用的文献

1
Combination of pleuroparenchymal fibroelastosis with non-specific interstitial pneumonia and bronchiolitis obliterans as a complication of hematopoietic stem cell transplantation - Clues to a potential mechanism.胸膜实质纤维弹性组织增生症合并非特异性间质性肺炎及闭塞性细支气管炎作为造血干细胞移植并发症——潜在机制线索
Respir Med Case Rep. 2019 Feb 4;26:244-247. doi: 10.1016/j.rmcr.2019.02.001. eCollection 2019.
2
Pleuroparenchymal fibroelastosis: a spectrum of histopathological and imaging phenotypes.胸膜肺弹力纤维增生症:一类具有组织病理学和影像学表型谱的疾病。
Eur Respir J. 2012 Aug;40(2):377-85. doi: 10.1183/09031936.00165111. Epub 2012 Mar 22.
3
Bronchiolitis obliterans syndrome (BOS), bronchiolitis obliterans organizing pneumonia (BOOP), and other late-onset noninfectious pulmonary complications following allogeneic hematopoietic stem cell transplantation.
闭塞性细支气管炎综合征(BOS)、机化性肺炎型闭塞性细支气管炎(BOOP)以及异基因造血干细胞移植后其他迟发性非感染性肺部并发症。
Biol Blood Marrow Transplant. 2007 Jul;13(7):749-59. doi: 10.1016/j.bbmt.2007.05.001.
4
National Institutes of Health Consensus Development Project on Criteria for Clinical Trials in Chronic Graft-versus-Host Disease: VI. Design of Clinical Trials Working Group report.美国国立卫生研究院慢性移植物抗宿主病临床试验标准共识发展项目:六、临床试验设计工作组报告。
Biol Blood Marrow Transplant. 2006 May;12(5):491-505. doi: 10.1016/j.bbmt.2006.03.004.
5
National Institutes of Health consensus development project on criteria for clinical trials in chronic graft-versus-host disease: I. Diagnosis and staging working group report.美国国立卫生研究院关于慢性移植物抗宿主病临床试验标准的共识发展项目:I. 诊断与分期工作组报告
Biol Blood Marrow Transplant. 2005 Dec;11(12):945-56. doi: 10.1016/j.bbmt.2005.09.004.