Department of Cardiology, Inselspital, Bern University Hospital (B.A., K.E.O., T.R.), University of Bern, Switzerland.
Cardiovascular and Clinical Academic Group, Molecular and Clinical Sciences Research Institute, St George's University of London, United Kingdom (A.A.).
Circulation. 2021 Nov 16;144(20):1646-1655. doi: 10.1161/CIRCULATIONAHA.121.055890. Epub 2021 Nov 15.
Arrhythmogenic cardiomyopathy (ACM) is a primary disease of the myocardium, predominantly caused by genetic defects in proteins of the cardiac intercalated disc, particularly, desmosomes. Transmission is mostly autosomal dominant with incomplete penetrance. ACM also has wide phenotype variability, ranging from premature ventricular contractions to sudden cardiac death and heart failure. Among other drivers and modulators of phenotype, inflammation in response to viral infection and immune triggers have been postulated to be an aggravator of cardiac myocyte damage and necrosis. This theory is supported by multiple pieces of evidence, including the presence of inflammatory infiltrates in more than two-thirds of ACM hearts, detection of different cardiotropic viruses in sporadic cases of ACM, the fact that patients with ACM often fulfill the histological criteria of active myocarditis, and the abundance of anti-desmoglein-2, antiheart, and anti-intercalated disk autoantibodies in patients with arrhythmogenic right ventricular cardiomyopathy. In keeping with the frequent familial occurrence of ACM, it has been proposed that, in addition to genetic predisposition to progressive myocardial damage, a heritable susceptibility to viral infections and immune reactions may explain familial clustering of ACM. Moreover, considerable in vitro and in vivo evidence implicates activated inflammatory signaling in ACM. Although the role of inflammation/immune response in ACM is not entirely clear, inflammation as a driver of phenotype and a potential target for mechanism-based therapy warrants further research. This review discusses the present evidence supporting the role of inflammatory and immune responses in ACM pathogenesis and proposes opportunities for translational and clinical investigation.
致心律失常性右室心肌病(ACM)是一种主要由心肌中细胞间桥盘蛋白,尤其是桥粒蛋白的遗传缺陷引起的原发性疾病。遗传方式主要为常染色体显性遗传,不完全外显。ACM 还具有广泛的表型变异性,从室性早搏到心脏性猝死和心力衰竭不等。在其他表型的驱动因素和调节因素中,病毒感染和免疫触发引起的炎症被认为是心肌细胞损伤和坏死的加重因素。这一理论得到了多方面证据的支持,包括超过三分之二的 ACM 心脏存在炎症浸润,散发性 ACM 病例中检测到不同的心脏病毒,ACM 患者常符合活动性心肌炎的组织学标准,以及抗桥粒蛋白-2、抗心和抗细胞间桥盘自身抗体在致心律失常性右室心肌病患者中的丰富存在。鉴于 ACM 常家族性发生,有人提出,除了遗传易感性导致进行性心肌损伤外,对病毒感染和免疫反应的遗传性易感性可能解释 ACM 的家族聚集性。此外,大量的体外和体内证据表明,激活的炎症信号在 ACM 中起作用。尽管炎症/免疫反应在 ACM 中的作用尚不完全清楚,但炎症作为表型的驱动因素和潜在的基于机制的治疗靶点值得进一步研究。本综述讨论了目前支持炎症和免疫反应在 ACM 发病机制中的作用的证据,并提出了转化和临床研究的机会。