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本文引用的文献

1
CT Angiography Findings of Pulmonary Arteriovenous Malformations in Children and Young Adults With Hereditary Hemorrhagic Telangiectasia.遗传性出血性毛细血管扩张症患儿和青年成人肺动静脉畸形的 CT 血管造影表现。
AJR Am J Roentgenol. 2020 Jun;214(6):1369-1376. doi: 10.2214/AJR.19.22012. Epub 2020 Apr 7.
2
Gender differences in hereditary hemorrhagic telangiectasia severity.遗传性出血性毛细血管扩张症严重程度的性别差异。
Orphanet J Rare Dis. 2020 Mar 2;15(1):63. doi: 10.1186/s13023-020-1337-5.
3
Bevacizumab for treating Hereditary Hemorrhagic Telangiectasia patients with severe hepatic involvement or refractory anemia.贝伐珠单抗治疗有严重肝受累或难治性贫血的遗传性出血性毛细血管扩张症患者。
PLoS One. 2020 Feb 7;15(2):e0228486. doi: 10.1371/journal.pone.0228486. eCollection 2020.
4
Treatment of Recurrent Pulmonary Arteriovenous Malformations: Comparison of Proximal Versus Distal Embolization Technique.复发性肺动静脉畸形的治疗:近端与远端栓塞技术比较。
Cardiovasc Intervent Radiol. 2020 Jan;43(1):29-36. doi: 10.1007/s00270-019-02328-0. Epub 2019 Aug 30.
5
Decreased levels of miR-28-5p and miR-361-3p and increased levels of insulin-like growth factor 1 mRNA in mononuclear cells from patients with hereditary hemorrhagic telangiectasia .遗传性出血性毛细血管扩张症患者单核细胞中 miR-28-5p 和 miR-361-3p 水平降低,胰岛素样生长因子 1 mRNA 水平升高。
Can J Physiol Pharmacol. 2019 Jun;97(6):562-569. doi: 10.1139/cjpp-2018-0508. Epub 2018 Dec 4.
6
Low serum haptoglobin and blood films suggest intravascular hemolysis contributes to severe anemia in hereditary hemorrhagic telangiectasia.低血清结合珠蛋白和血涂片提示血管内溶血导致遗传性出血性毛细血管扩张症中的严重贫血。
Haematologica. 2019 Apr;104(4):e127-e130. doi: 10.3324/haematol.2018.205682. Epub 2018 Oct 18.
7
British Thoracic Society Clinical Statement on Pulmonary Arteriovenous Malformations.英国胸科学会肺动静脉畸形临床声明。
Thorax. 2017 Dec;72(12):1154-1163. doi: 10.1136/thoraxjnl-2017-210764.
8
Pulmonary arteriovenous malformations and their mimics.肺动静脉畸形及其类似病变
Clin Radiol. 2015 Jan;70(1):96-110. doi: 10.1016/j.crad.2014.09.003. Epub 2014 Nov 17.
9
Treated pulmonary arteriovenous malformations: patterns of persistence and associated retreatment success.治疗后的肺动静脉畸形:持续性模式和相关的再治疗成功率。
Radiology. 2013 Dec;269(3):919-26. doi: 10.1148/radiol.13122153. Epub 2013 Oct 28.
10
Enlargement of the bronchial arteries, and their anastomoses with the pulmonary arteries in bronchiectasis.支气管扩张症中支气管动脉的增粗及其与肺动脉的吻合。
Am J Pathol. 1949 Mar;25(2):211-31.

遗传性出血性毛细血管扩张症和肺动静脉畸形。

Hereditary haemorrhagic telangiectasia and pulmonary arteriovenous malformations.

机构信息

Department of Acute Medicine, The Royal Berkshire Hospital, Reading, UK

Department of Acute Medicine, The Royal Berkshire Hospital, Reading, UK.

出版信息

BMJ Case Rep. 2021 Jan 8;14(1):e238385. doi: 10.1136/bcr-2020-238385.

DOI:10.1136/bcr-2020-238385
PMID:33419752
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7799076/
Abstract

Hereditary haemorrhagic telangiectasia (HHT) also known as Osler-Weber-Rendu syndrome is an autosomal dominant disorder affecting 1 in 8000 individuals. The eponym recognises the 19th-century physicians William Osler, Henri Jules Louis Marie Rendu and Frederick Parkes Weber who each independently described the disease. It is characterised by epistaxis, telangiectasia and visceral arteriovenous malformations. Individuals with HHT have been found to have abnormal plasma concentrations of transforming growth factor beta and vascular endothelial growth factor secondary to mutations in ENG, ACVRL1 and MADH4. Pulmonary artery malformations (PAVMs) are abnormal communications between pulmonary arteries and veins and are found in up to 50% of individuals with HHT. The clinical features suggestive of PAVMs are stigmata of right to left shunting such as dyspnoea, hypoxaemia, cyanosis, cerebral embolism and unexplained haemoptysis or haemothorax. The authors present the case of a 33-year-old woman presenting with progressive dyspnoea during the COVID-19 pandemic. She had a typical presentation of HHT with recurrent epistaxis, telangiectasia and pulmonary arteriovenous malformations. Although rare, PAVM should be considered in individuals presenting to the emergency department with dyspnoea and hypoxaemia. Delayed diagnosis can result in fatal embolic and haemorrhagic complications.

摘要

遗传性出血性毛细血管扩张症(HHT),又称 Osler-Weber-Rendu 综合征,是一种常染色体显性遗传疾病,影响每 8000 人中的 1 人。这个名字是为了纪念 19 世纪的三位医生:William Osler、Henri Jules Louis Marie Rendu 和 Frederick Parkes Weber,他们各自独立地描述了这种疾病。其特征为鼻出血、毛细血管扩张和内脏动静脉畸形。患有 HHT 的个体由于 ENG、ACVRL1 和 MADH4 的突变,导致转化生长因子β和血管内皮生长因子的血浆浓度异常。肺动脉畸形(PAVM)是指肺动脉和静脉之间的异常沟通,在多达 50%的 HHT 患者中发现。提示 PAVM 的临床特征包括右向左分流的迹象,如呼吸困难、低氧血症、发绀、脑栓塞和不明原因的咯血或血胸。作者报告了一例 33 岁女性在 COVID-19 大流行期间出现进行性呼吸困难的病例。她有典型的 HHT 表现,包括反复鼻出血、毛细血管扩张和肺动脉动静脉畸形。尽管罕见,但在因呼吸困难和低氧血症就诊急诊的个体中,应考虑 PAVM。延迟诊断可导致致命的栓塞和出血并发症。