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本文引用的文献

1
Case 11-2018: A 48-Year-Old Woman with Recurrent Venous Thromboembolism and Pulmonary Artery Aneurysm.病例11 - 2018:一名48岁复发性静脉血栓栓塞症和肺动脉瘤女性患者。
N Engl J Med. 2018 Apr 12;378(15):1430-1438. doi: 10.1056/NEJMcpc1800323.
2
Acquired pulmonary artery pseudoaneurysms: a pictorial review.获得性肺动脉假性动脉瘤:影像学综述
Br J Radiol. 2017 May;90(1073):20160783. doi: 10.1259/bjr.20160783. Epub 2017 Mar 24.
3
Moderate hemoptysis caused by hughes-stovin syndrome.休斯-斯托文综合征所致的中度咯血。
Clin Pract. 2014 Nov 6;4(3):647. doi: 10.4081/cp.2014.647. eCollection 2014 Oct 30.
4
Pulmonary artery aneurysms: four case reports and literature review.肺动脉瘤:四例病例报告及文献综述
Int J Angiol. 2013 Sep;22(3):143-8. doi: 10.1055/s-0033-1347907.
5
Behcet's disease with major vascular involvement.伴有主要血管受累的白塞病。
BMJ Case Rep. 2013 Nov 8;2013:bcr2013200893. doi: 10.1136/bcr-2013-200893.
6
Hughes-Stovin syndrome.休斯-斯托文综合征。
Orphanet J Rare Dis. 2011 Apr 13;6:15. doi: 10.1186/1750-1172-6-15.
7
Pulmonary artery pseudoaneurysm: etiology, presentation, diagnosis, and treatment.肺动脉假性动脉瘤:病因、表现、诊断及治疗
Semin Intervent Radiol. 2007 Mar;24(1):119-23. doi: 10.1055/s-2007-971202.
8
The revised Ghent nosology for the Marfan syndrome.修订版马凡综合征根特分类法。
J Med Genet. 2010 Jul;47(7):476-85. doi: 10.1136/jmg.2009.072785.
9
Hughes-Stovin syndrome: a case report and review of the literature.休斯-斯托文综合征:一例病例报告及文献综述
Cases J. 2009 Jan 29;2:98. doi: 10.1186/1757-1626-2-98.
10
Pulmonary artery aneurysms in Behçet syndrome.白塞综合征中的肺动脉瘤
Am J Med. 2004 Dec 1;117(11):867-70. doi: 10.1016/j.amjmed.2004.05.027.

休-斯托文综合征:“不完全型白塞病”。一名患有复发性肺栓塞和肺动脉瘤的年轻成年人的病例研究。

Hugh-Stovin syndrome: the 'incomplete Behcet's disease'. A case study of a young adult with recurrent pulmonary embolism and pulmonary arterial aneurysms.

作者信息

Ukemenam Chinedu, Muppaneni Sivaram Prabhuji, De Debapriya, Lacasse Alexandre

机构信息

Department of Internal Medicine, SSM Saint Mary's Hospital, Saint Louis, Missouri, USA.

出版信息

J Community Hosp Intern Med Perspect. 2021 Jun 21;11(4):566-567. doi: 10.1080/20009666.2020.1816273.

DOI:10.1080/20009666.2020.1816273
PMID:34211671
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8221138/
Abstract

Hugh-Stovin Syndrome (HSS) is characterized by recurrent thrombophlebitis and multiple pulmonary and/or bronchial artery aneurysms indistinguishable from the cardiovascular features seen in Behcet's disease (BD). Our case describes a 30-year-old male with recurrent pulmonary embolism and bilateral pulmonary aneurysms. Autoimmune, hypercoagulable, and infectious work up were negative. Elevated inflammatory markers and absence of the typical clinical findings seen in BD led to the diagnosis of Hugh-Stovin syndrome (HSS). Immunosuppression using steroids and azathioprine led to clinical response. Anticoagulation was continued based on risk/benefit ratio.

摘要

休-斯托文综合征(HSS)的特征为复发性血栓性静脉炎以及多发性肺和/或支气管动脉动脉瘤,其心血管特征与白塞病(BD)难以区分。我们的病例描述了一名30岁男性,患有复发性肺栓塞和双侧肺动脉瘤。自身免疫、高凝状态及感染相关检查均为阴性。炎症标志物升高且缺乏BD的典型临床表现,从而诊断为休-斯托文综合征(HSS)。使用类固醇和硫唑嘌呤进行免疫抑制治疗后出现临床反应。根据风险/获益比继续进行抗凝治疗。