From the Department of Neurology (K.K.T., C.D.S.), University of Minnesota, Minneapolis; Department of Neurology (S.D.P., J.L.S.), Ronald Reagan UCLA Medical Center, Los Angeles, CA; and Department of Clinical and Molecular Medicine (C.L.S.), Imperial College London, UK.
Neurology. 2022 Feb 1;98(5):188-198. doi: 10.1212/WNL.0000000000013169. Epub 2021 Dec 8.
The potential of covert pulmonary arteriovenous malformations (PAVMs) to cause early onset, preventable ischemic strokes is not well known to neurologists. This is evident by their lack of mention in serial American Heart Association/American Stroke Association (AHA/ASA) Guidelines and the single case report biased literature of recent years. We performed PubMed and Cochrane database searches for major studies on ischemic stroke and PAVMs published from January 1, 1974, through April 3, 2021. This identified 24 major observational studies, 3 societal guidelines, 1 nationwide analysis, 3 systematic reviews, 21 other review/opinion articles, and 18 recent (2017-2021) case reports/series that were synthesized. Key points are that patients with PAVMs have ischemic stroke a decade earlier than routine stroke, losing 9 extra healthy life-years per patient in the recent US nationwide analysis (2005-2014). Large-scale thoracic CT screens of the general population in Japan estimate PAVM prevalence to be 38/100,000 (95% confidence interval 18-76), with ischemic stroke rates exceeding 10% across PAVM series dating back to the 1950s, with most PAVMs remaining undiagnosed until the time of clinical stroke. Notably, the rate of PAVM diagnoses doubled in US ischemic stroke hospitalizations between 2005 and 2014. The burden of silent cerebral infarction approximates to twice that of clinical stroke. More than 80% of patients have underlying hereditary hemorrhagic telangiectasia. The predominant stroke mechanism is paradoxical embolization of platelet-rich emboli, with iron deficiency emerging as a modifiable risk factor. PAVM-related ischemic strokes may be cortical or subcortical, but very rarely cause proximal large vessel occlusions. Single antiplatelet therapy may be effective for secondary stroke prophylaxis, with dual antiplatelet or anticoagulation therapy requiring nuanced risk-benefit analysis given their risk of aggravating iron deficiency. This review summarizes the ischemic stroke burden from PAVMs, the implicative pathophysiology, and relevant diagnostic and treatment overviews to facilitate future incorporation into AHA/ASA guidelines.
神经科医生对隐匿性肺动静脉畸形(PAVM)导致早期、可预防的缺血性卒中的潜在风险认识不足。这从他们在连续的美国心脏协会/美国卒中协会(AHA/ASA)指南中没有提及,以及近年来的单一病例报告偏倚文献中可以明显看出。我们在 PubMed 和 Cochrane 数据库中检索了 1974 年 1 月 1 日至 2021 年 4 月 3 日期间发表的关于缺血性卒中和 PAVM 的主要研究。这确定了 24 项主要观察性研究、3 项社会指南、1 项全国性分析、3 项系统评价、21 篇其他综述/观点文章和 18 篇最近(2017-2021 年)的病例报告/系列,对这些文章进行了综合分析。关键要点是,患有 PAVM 的患者比常规卒中早 10 年发生缺血性卒中,最近在美国全国性分析(2005-2014 年)中每位患者损失 9 个额外的健康寿命年。日本对普通人群进行大规模的胸部 CT 筛查,估计 PAVM 的患病率为 38/100,000(95%置信区间 18-76),自 20 世纪 50 年代以来,多项 PAVM 系列研究显示缺血性卒中超 10%,大多数 PAVM 直到发生临床卒中时才被诊断出来。值得注意的是,2005 年至 2014 年期间,美国缺血性卒中住院患者的 PAVM 诊断率增加了一倍。无症状性脑梗死的负担与临床卒中相当。超过 80%的患者存在遗传性出血性毛细血管扩张症。主要的卒中机制是富含血小板的栓子的反常栓塞,而铁缺乏症作为一个可改变的危险因素正在出现。PAVM 相关的缺血性卒中可能是皮质或皮质下的,但很少导致近端大血管闭塞。单一抗血小板治疗可能对二级卒中预防有效,双重抗血小板或抗凝治疗需要进行细致的风险效益分析,因为它们有加重铁缺乏症的风险。本综述总结了 PAVM 引起的缺血性卒中负担、潜在的病理生理学以及相关的诊断和治疗概述,以促进未来纳入 AHA/ASA 指南。