Department of Acute Medicine, The Royal Berkshire Hospital, Reading, UK
Department of Acute Medicine, The Royal Berkshire Hospital, Reading, UK.
BMJ Case Rep. 2021 Jan 8;14(1):e238385. doi: 10.1136/bcr-2020-238385.
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.
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。