Huang Lei, Tang Shuangbo, Chen Yili, Zhang Liyong, Yin Kun, Wu Yeda, Zheng Jinxiang, Wu Qiuping, Makielski Jonathan C, Cheng Jianding
Department of Forensic Pathology, Zhongshan School of Medicine, Sun Yat-sen University, Guangzhou, Guangdong, China.
Department of Cardiology, The First Affiliated Hospital of Sun Yat-sen University, Guangzhou, Guangdong, China.
Int J Legal Med. 2017 May;131(3):621-628. doi: 10.1007/s00414-016-1516-z. Epub 2016 Dec 28.
Sudden unexplained nocturnal death syndrome (SUNDS) is a perplexing disorder to both forensic pathologists and clinic physicians. Clinical features of SUNDS survivors suggested that SUNDS is similar to Brugada syndrome (BrS). Leucine-rich repeat containing 10 (LRRC10) gene was a newly identified gene linked to dilated cardiomyopathy, a disease associated with sudden cardiac death. To investigate the prevalence and spectrum of genetic variants of LRRC10 gene in SUNDS and BrS, the coding regions of LRRC10 were genetically screened in 113 sporadic SUNDS victims (from January 2005 to December 2015, 30.7 ± 7.5 years) and ten BrS patients (during January 2010 to December 2014, 38.7 ± 10.3 years) using direct Sanger sequencing. Afterwards, LRRC10 missense variant carriers were screened for a panel of 80 genes known to be associated with inherited cardiac arrhythmia/cardiomyopathy using target-captured next-generation sequencing. In this study, an in silico-predicted malignant LRRC10 mutation p.E129K was detected in one SUNDS victim without pathogenic rare variant in a panel of 80 arrhythmia/cardiomyopathy-related genes. We also provided evidence to show that rare variant p.P69L might contribute to the genetic cause for one SUNDS victim and two BrS family members. This is the first report of genetic screening of LRRC10 in Chinese SUNDS victims and BrS patients. LRRC10 may be a new susceptible gene for SUNDS, and LRRC10 variant was initially and genetically linked to BrS-associated arrhythmia.
不明原因夜间猝死综合征(SUNDS)对于法医病理学家和临床医生而言都是一种令人困惑的病症。SUNDS幸存者的临床特征表明,SUNDS与Brugada综合征(BrS)相似。富含亮氨酸重复序列10(LRRC10)基因是一个新发现的与扩张型心肌病相关的基因,扩张型心肌病是一种与心源性猝死相关的疾病。为了研究LRRC10基因在SUNDS和BrS中的遗传变异流行情况及范围,我们采用直接Sanger测序法,对113例散发性SUNDS受害者(2005年1月至2015年12月,年龄30.7±7.5岁)和10例BrS患者(2010年1月至2014年12月,年龄38.7±10.3岁)的LRRC10编码区进行了基因筛查。之后,利用靶向捕获二代测序技术,对一组已知与遗传性心律失常/心肌病相关的80个基因,筛查LRRC10错义变异携带者。在本研究中,在一名SUNDS受害者中检测到一个计算机预测的恶性LRRC10突变p.E129K,而在一组80个与心律失常/心肌病相关的基因中未发现致病罕见变异。我们还提供证据表明,罕见变异p.P69L可能是一名SUNDS受害者和两名BrS家族成员的遗传病因。这是中国SUNDS受害者和BrS患者中LRRC10基因筛查的首份报告。LRRC10可能是SUNDS的一个新的易感基因,并且LRRC10变异最初在基因层面与BrS相关心律失常有关。