Suppr超能文献

中国汉族人群不明原因夜间猝死综合征中LRRC10的分子病理学研究

Molecular pathological study on LRRC10 in sudden unexplained nocturnal death syndrome in the Chinese Han population.

作者信息

Huang Lei, Tang Shuangbo, Chen Yili, Zhang Liyong, Yin Kun, Wu Yeda, Zheng Jinxiang, Wu Qiuping, Makielski Jonathan C, Cheng Jianding

机构信息

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.

出版信息

Int J Legal Med. 2017 May;131(3):621-628. doi: 10.1007/s00414-016-1516-z. Epub 2016 Dec 28.

Abstract

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.

摘要

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。

相似文献

1
Molecular pathological study on LRRC10 in sudden unexplained nocturnal death syndrome in the Chinese Han population.
Int J Legal Med. 2017 May;131(3):621-628. doi: 10.1007/s00414-016-1516-z. Epub 2016 Dec 28.
2
Association of common and rare variants of SCN10A gene with sudden unexplained nocturnal death syndrome in Chinese Han population.
Int J Legal Med. 2017 Jan;131(1):53-60. doi: 10.1007/s00414-016-1397-1. Epub 2016 Jun 7.
3
Identification of rare variants of DSP gene in sudden unexplained nocturnal death syndrome in the southern Chinese Han population.
Int J Legal Med. 2016 Mar;130(2):317-22. doi: 10.1007/s00414-015-1275-2. Epub 2015 Nov 19.
5
Postmortem genetic screening of SNPs in RyR2 gene in sudden unexplained nocturnal death syndrome in the southern Chinese Han population.
Forensic Sci Int. 2014 Feb;235:14-8. doi: 10.1016/j.forsciint.2013.12.007. Epub 2013 Dec 16.
6
Postmortem molecular analysis of KCNQ1, KCNH2, KCNE1 and KCNE2 genes in sudden unexplained nocturnal death syndrome in the Chinese Han population.
Forensic Sci Int. 2013 Sep 10;231(1-3):82-7. doi: 10.1016/j.forsciint.2013.04.020. Epub 2013 May 15.
7
Is sudden unexplained nocturnal death syndrome in Southern China a cardiac sodium channel dysfunction disorder?
Forensic Sci Int. 2014 Mar;236:38-45. doi: 10.1016/j.forsciint.2013.12.033. Epub 2014 Jan 7.
9
Molecular Autopsy of Desmosomal Protein Plakophilin-2 in Sudden Unexplained Nocturnal Death Syndrome.
J Forensic Sci. 2016 May;61(3):687-91. doi: 10.1111/1556-4029.13027. Epub 2016 Jan 4.
10
HCN4 Gene Variations in Sudden Unexplained Nocturnal Death Syndrome in the Southern Han Chinese Population.
J Forensic Sci. 2019 Jul;64(4):1112-1118. doi: 10.1111/1556-4029.13958. Epub 2018 Nov 19.

引用本文的文献

1
Genetic Basis of Brugada Syndrome.
Biomedicines. 2025 Jul 16;13(7):1740. doi: 10.3390/biomedicines13071740.
2
A genetically encoded actuator boosts L-type calcium channel function in diverse physiological settings.
Sci Adv. 2024 Nov;10(44):eadq3374. doi: 10.1126/sciadv.adq3374. Epub 2024 Oct 30.
3
Cardiac L-type calcium channel regulation by Leucine-Rich Repeat-Containing Protein 10.
Channels (Austin). 2024 Dec;18(1):2355121. doi: 10.1080/19336950.2024.2355121. Epub 2024 May 19.
4
Brugada Syndrome: From Molecular Mechanisms and Genetics to Risk Stratification.
Int J Mol Sci. 2023 Feb 7;24(4):3328. doi: 10.3390/ijms24043328.
5
Brugada Syndrome: Oligogenic or Mendelian Disease?
Int J Mol Sci. 2020 Mar 1;21(5):1687. doi: 10.3390/ijms21051687.
6
Sudden Unexplained Nocturnal Death Syndrome: The Hundred Years' Enigma.
J Am Heart Assoc. 2018 Mar 3;7(5):e007837. doi: 10.1161/JAHA.117.007837.

本文引用的文献

1
Association of common and rare variants of SCN10A gene with sudden unexplained nocturnal death syndrome in Chinese Han population.
Int J Legal Med. 2017 Jan;131(1):53-60. doi: 10.1007/s00414-016-1397-1. Epub 2016 Jun 7.
2
A novel conditional mouse model for Nkx2-5 reveals transcriptional regulation of cardiac ion channels.
Differentiation. 2016 Jan-Mar;91(1-3):29-41. doi: 10.1016/j.diff.2015.12.003. Epub 2016 Feb 17.
3
SCN5A(K817E), a novel Brugada syndrome-associated mutation that alters the activation gating of NaV1.5 channel.
Heart Rhythm. 2016 May;13(5):1113-1120. doi: 10.1016/j.hrthm.2016.01.008. Epub 2016 Jan 8.
4
LRRC10 is required to maintain cardiac function in response to pressure overload.
Am J Physiol Heart Circ Physiol. 2016 Jan 15;310(2):H269-78. doi: 10.1152/ajpheart.00717.2014. Epub 2015 Nov 25.
5
Identification of rare variants of DSP gene in sudden unexplained nocturnal death syndrome in the southern Chinese Han population.
Int J Legal Med. 2016 Mar;130(2):317-22. doi: 10.1007/s00414-015-1275-2. Epub 2015 Nov 19.
6
Prevalence and spectrum of LRRC10 mutations associated with idiopathic dilated cardiomyopathy.
Mol Med Rep. 2015 Sep;12(3):3718-3724. doi: 10.3892/mmr.2015.3843. Epub 2015 May 25.
7
A case-control study of sudden unexplained nocturnal death syndrome in the southern Chinese Han population.
Am J Forensic Med Pathol. 2015 Mar;36(1):39-43. doi: 10.1097/PAF.0000000000000135.
8
Complex Brugada syndrome inheritance in a family harbouring compound SCN5A and CACNA1C mutations.
Basic Res Cardiol. 2014;109(6):446. doi: 10.1007/s00395-014-0446-5. Epub 2014 Oct 24.
9
SCN5A-related dilated cardiomyopathy: what do we know?
Heart Rhythm. 2014 Aug;11(8):1454-5. doi: 10.1016/j.hrthm.2014.05.031. Epub 2014 May 29.
10
Novel SCN5A mutation in amiodarone-responsive multifocal ventricular ectopy-associated cardiomyopathy.
Heart Rhythm. 2014 Aug;11(8):1446-53. doi: 10.1016/j.hrthm.2014.04.042. Epub 2014 May 9.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验