University of Cambridge School of Clinical Medicine, Addenbrooke's and Papworth Hospitals, Cambridge, UK.
Indiana University School of Medicine, Indianapolis, IN, USA.
Eur Respir J. 2019 Jan 24;53(1). doi: 10.1183/13993003.01899-2018. Print 2019 Jan.
Since 2000 there have been major advances in our understanding of the genetic and genomics of pulmonary arterial hypertension (PAH), although there remains much to discover. Based on existing knowledge, around 25-30% of patients diagnosed with idiopathic PAH have an underlying Mendelian genetic cause for their condition and should be classified as heritable PAH (HPAH). Here, we summarise the known genetic and genomic drivers of PAH, the insights these provide into pathobiology, and the opportunities afforded for development of novel therapeutic approaches. In addition, factors determining the incomplete penetrance observed in HPAH are discussed. The currently available approaches to genetic testing and counselling, and the impact of a genetic diagnosis on clinical management of the patient with PAH, are presented. Advances in DNA sequencing technology are rapidly expanding our ability to undertake genomic studies at scale in large cohorts. In the future, such studies will provide a more complete picture of the genetic contribution to PAH and, potentially, a molecular classification of this disease.
自 2000 年以来,我们对肺动脉高压(PAH)的遗传和基因组学有了重大的认识进展,尽管仍有许多需要发现。基于现有知识,约 25-30%被诊断为特发性 PAH 的患者存在导致其病情的潜在孟德尔遗传原因,应被归类为遗传性 PAH(HPAH)。在这里,我们总结了已知的 PAH 的遗传和基因组驱动因素,这些因素为病理生物学提供了哪些见解,以及为开发新型治疗方法提供了哪些机会。此外,还讨论了导致 HPAH 中观察到不完全外显率的因素。目前可用的遗传测试和咨询方法,以及遗传诊断对 PAH 患者临床管理的影响也被呈现出来。DNA 测序技术的进步正在迅速提高我们在大规模的大型队列中进行基因组研究的能力。在未来,此类研究将更全面地了解遗传对 PAH 的贡献,并可能为这种疾病提供一种分子分类。