Miyata Kazuaki, Ohno Seiko, Itoh Hideki, Horie Minoru
Department of Cardiovascular and Respiratory Medicine, Shiga University of Medical Science, Japan.
Department of Cardiovascular Medicine, Iga City General Hospital, Japan.
Intern Med. 2018 Jul 1;57(13):1813-1817. doi: 10.2169/internalmedicine.9843-17. Epub 2018 Feb 9.
Objective Catecholaminergic polymorphic ventricular tachycardia (CPVT) is a lethal inherited disease characterized by ventricular arrhythmias induced by physical exercise or emotional stress. The major cause of CPVT is mutations in RYR2, which encodes the cardiac ryanodine receptor channel. Recent advances in sequencing technology have yielded incidental findings of RYR2 variants in other cardiac diseases. Analyzing the characteristics of RYR2 variants related to CPVT will be useful for differentiation from those related to other cardiac diseases. We examined the phenotypic characteristics of patients with RYR2 variants. Methods Seventy-nine probands carrying RYR2 variants whose diagnoses were either CPVT (n=68) or long QT syndrome (LQTS; n=11) were enrolled. We compared the characteristics of the electrocardiogram (ECG) and the location of the RYR2 mutations-N-terminal (NT), central region (CR) or C-terminal (CT)-between the two patient groups. Results Using the ECGs available from 53 probands before β-blocker therapies, we analyzed the heart rates (HRs). CPVT probands showed bradycardia more frequently (25/44; 57%) than LQTS probands (1/9; 11%; p=0.024). In CPVT patients, 20 mutations were located in NT, 25 in CR and 23 in CT. In LQTS patients, 5 mutations were located in NT, 2 in CR and 4 in CT. There were no significant differences in the locations of the RYR2 mutations between the phenotypes. Conclusion Bradycardia was highly correlated with the phenotype of CPVT. When a clinically-diagnosed LQTS patient with bradycardia carries an RYR2 mutation, we should be careful to avoid making a misdiagnosis, as the patient may actually have CPVT.
目的 儿茶酚胺能多形性室性心动过速(CPVT)是一种致命的遗传性疾病,其特征为体育锻炼或情绪应激诱发的室性心律失常。CPVT的主要病因是编码心脏雷诺丁受体通道的RYR2基因突变。测序技术的最新进展在其他心脏疾病中偶然发现了RYR2变异。分析与CPVT相关的RYR2变异特征将有助于与其他心脏疾病相关的变异进行区分。我们研究了携带RYR2变异患者的表型特征。方法 纳入79例携带RYR2变异的先证者,其诊断为CPVT(n = 68)或长QT综合征(LQTS;n = 11)。我们比较了两组患者的心电图(ECG)特征以及RYR2突变的位置——N端(NT)、中央区域(CR)或C端(CT)。结果 利用53例先证者在β受体阻滞剂治疗前的心电图,我们分析了心率(HR)。CPVT先证者出现心动过缓的频率(25/44;57%)高于LQTS先证者(1/9;11%;p = 0.024)。在CPVT患者中,20个突变位于NT,25个位于CR,23个位于CT。在LQTS患者中,5个突变位于NT,2个位于CR,4个位于CT。两种表型的RYR2突变位置无显著差异。结论 心动过缓与CPVT表型高度相关。当临床诊断为LQTS且伴有心动过缓的患者携带RYR2突变时,我们应谨慎避免误诊,因为该患者实际上可能患有CPVT。