Langleben David, Orfanos Stylianos
1 Center for Pulmonary Vascular Disease, Division of Cardiology, Jewish General Hospital, McGill University, Montreal, Quebec Canada.
2 Pulmonary Hypertension Clinic, Department of Critical Care, Attikon Hospital, National and Kapodistirian University of Athens, Athens, Greece.
Pulm Circ. 2017 Jul-Sep;7(3):588-597. doi: 10.1177/2045893217714231. Epub 2017 Jun 20.
Within the cohort of patients suffering from idiopathic pulmonary arterial hypertension (IPAH) is a group that responds dramatically (VR-PAH) to an acute vasodilator challenge and that has excellent long-term hemodynamic improvement and prognosis on high dose calcium channel blockers compared with vasodilator non-responders (VN-PAH). For the purposes of diagnosing VR-PAH, there is to date no test to replace the acute vasodilator challenge. However, recent studies have identified markers that may aid in the identification of VR-PAH, including peripheral blood lymphocyte RNA expression levels of desmogelin-2 and Ras homolog gene family member Q, and plasma levels of provirus integration site for Moloney murine leukemia virus. Genome wide-array studies of peripheral blood DNA have demonstrated differences in disease specific genetic variants between VR-PAH and NR-PAH, with particular convergence on cytoskeletal function pathways and Wnt signaling pathways. These studies offer hope for future non-invasive identification of VR-PAH, and insights into pathogenesis that may lead to novel therapies. Examination of the degree of pulmonary microvascular perfusion in PAH has offered additional insights. During the acute vasodilator challenge, VR-PAH patients demonstrate true vasodilation with recruitment and increased perfusion of the capillary bed, while VN-PAH patients are unable to recruit vasculature. In the very few reports of lung histology, VR-PAH has more medial thickening in the precapillary arterioles, while VN-PAH has the classic histology of PAH, including intimal thickening. VR-PAH is a disorder with a phenotype distinct from VN-PAH and other types of PAH, and should be considered separately in the classification of PAH.
在特发性肺动脉高压(IPAH)患者队列中,有一组患者对急性血管扩张剂激发试验有显著反应(VR-PAH),与血管扩张剂无反应者(VN-PAH)相比,他们在高剂量钙通道阻滞剂治疗下有出色的长期血流动力学改善和预后。就诊断VR-PAH而言,迄今为止尚无测试可替代急性血管扩张剂激发试验。然而,最近的研究已经确定了可能有助于识别VR-PAH的标志物,包括外周血淋巴细胞中去桥粒芯糖蛋白-2和Ras同源基因家族成员Q的RNA表达水平,以及莫洛尼鼠白血病病毒前病毒整合位点的血浆水平。外周血DNA的全基因组阵列研究表明,VR-PAH和NR-PAH之间存在疾病特异性基因变异差异,尤其集中在细胞骨架功能途径和Wnt信号通路。这些研究为未来无创识别VR-PAH带来了希望,并为可能导致新疗法的发病机制提供了见解。对PAH中肺微血管灌注程度的检查提供了更多见解。在急性血管扩张剂激发试验期间,VR-PAH患者表现出真正的血管扩张,伴有毛细血管床的募集和灌注增加,而VN-PAH患者无法募集血管。在极少数肺组织学报告中,VR-PAH在毛细血管前小动脉中有更多的中层增厚,而VN-PAH具有PAH的典型组织学特征,包括内膜增厚。VR-PAH是一种表型不同于VN-PAH和其他类型PAH的疾病,在PAH分类中应单独考虑。