Skinner Jonathan R, Winbo Annika, Abrams Dominic, Vohra Jitendra, Wilde Arthur A
The Cardiac Inherited Disease Group, Auckland, New Zealand; Greenlane Paediatric and Congenital Cardiac Services, Starship Children's Hospital, Auckland, New Zealand; Department of Paediatrics, Child and Youth Health, University of Auckland, New Zealand.
The Cardiac Inherited Disease Group, Auckland, New Zealand; Greenlane Paediatric and Congenital Cardiac Services, Starship Children's Hospital, Auckland, New Zealand; Department of Neurophysiology, University of Auckland, New Zealand.
Heart Lung Circ. 2019 Jan;28(1):22-30. doi: 10.1016/j.hlc.2018.09.007. Epub 2018 Oct 4.
Forty per cent (40%) of sudden unexpected natural deaths in people under 35 years of age are associated with a negative autopsy, and the cardiac ion channelopathies are the prime suspects in such cases. Long QT syndrome (LQTS), Brugada syndrome (BrS) and catecholaminergic polymorphic ventricular tachycardia (CPVT) are the most commonly identified with genetic testing. The cellular action potential driving the heart cycle is shaped by a specific series of depolarising and repolarising ion currents mediated by ion channels. Alterations in any of these currents, and in the availability of intracellular free calcium, leaves the myocardium vulnerable to polymorphic ventricular tachycardia or ventricular fibrillation. Each channelopathy has its own electrocardiogram (ECG) signature, typical mode of presentation, and most commonly related gene. Long QT type 1 (gene, KCNQ1) and CPVT (gene, RyR2) typically present with cardiac events (ie syncope or cardiac arrest) during or immediately after exercise in young males; long QT type 2 (gene, KCNH2) after startle or during the night in adult females-particularly early post-partum, and long QT type 3 and Brugada syndrome (gene, SCN5A) during the night in young adult males. They are commonly misdiagnosed as seizure disorders. Fever-triggered cardiac events should also raise the suspicion of BrS. This review summarises genetics, cellular mechanisms, risk stratification and treatments. Beta blockers are the mainstay of treatment for long QT syndrome and CPVT, and flecainide is remarkably effective in CPVT. Brugada syndrome is genetically a more complex disease than the others, and risk stratification and management is more difficult.
35岁以下人群中,40%的意外自然猝死尸检呈阴性,心脏离子通道病是这类病例的主要怀疑对象。长QT综合征(LQTS)、Brugada综合征(BrS)和儿茶酚胺能多形性室性心动过速(CPVT)是基因检测中最常发现的疾病。驱动心脏周期的细胞动作电位由离子通道介导的一系列特定的去极化和复极化离子电流形成。这些电流中的任何一种发生改变,以及细胞内游离钙的可用性发生改变,都会使心肌易患多形性室性心动过速或心室颤动。每种通道病都有其自身的心电图(ECG)特征、典型的表现方式以及最常相关的基因。长QT1型(基因,KCNQ1)和CPVT(基因,RyR2)通常在年轻男性运动期间或运动后立即出现心脏事件(即晕厥或心脏骤停);长QT2型(基因,KCNH2)在成年女性受到惊吓后或夜间出现,尤其是产后早期,长QT3型和Brugada综合征(基因,SCN5A)在年轻成年男性夜间出现。它们通常被误诊为癫痫疾病。发热引发的心脏事件也应引起对Brugada综合征的怀疑。本综述总结了遗传学、细胞机制、风险分层和治疗方法。β受体阻滞剂是长QT综合征和CPVT治疗的主要药物,氟卡尼对CPVT非常有效。Brugada综合征在遗传上比其他疾病更复杂,风险分层和管理也更困难。