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Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection

Total anomalous pulmonary venous connection (TAPVC) is a rare congenital heart anomaly. Several genes have been associated TAPVC but the mechanisms remain elusive. To search novel CNVs and candidate genes, we screened a cohort of 78 TAPVC cases and 100 healthy controls for rare copy number variants...

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Autores principales: Shi, Xin, Cheng, Liangping, Jiao, XianTing, Chen, Bo, Li, Zixiong, Liang, Yulai, Liu, Wei, Wang, Jing, Liu, Gang, Xu, Yuejuan, Sun, Jing, Fu, Qihua, Lu, Yanan, Chen, Sun
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6265481/
https://www.ncbi.nlm.nih.gov/pubmed/30532766
http://dx.doi.org/10.3389/fgene.2018.00559
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author Shi, Xin
Cheng, Liangping
Jiao, XianTing
Chen, Bo
Li, Zixiong
Liang, Yulai
Liu, Wei
Wang, Jing
Liu, Gang
Xu, Yuejuan
Sun, Jing
Fu, Qihua
Lu, Yanan
Chen, Sun
author_facet Shi, Xin
Cheng, Liangping
Jiao, XianTing
Chen, Bo
Li, Zixiong
Liang, Yulai
Liu, Wei
Wang, Jing
Liu, Gang
Xu, Yuejuan
Sun, Jing
Fu, Qihua
Lu, Yanan
Chen, Sun
author_sort Shi, Xin
collection PubMed
description Total anomalous pulmonary venous connection (TAPVC) is a rare congenital heart anomaly. Several genes have been associated TAPVC but the mechanisms remain elusive. To search novel CNVs and candidate genes, we screened a cohort of 78 TAPVC cases and 100 healthy controls for rare copy number variants (CNVs) using whole exome sequencing (WES). Then we identified pathogenic CNVs by statistical comparisons between case and control groups. After that, we identified altogether eight pathogenic CNVs of seven candidate genes (PCSK7, RRP7A, SERHL, TARP, TTN, SERHL2, and NBPF3). All these seven genes have not been described previously to be related to TAPVC. After network analysis of these candidate genes and 27 known pathogenic genes derived from the literature and publicly database, PCSK7 and TTN were the most important genes for TAPVC than other genes. Our study provides novel candidate genes potentially related to this rare congenital birth defect (CHD) which should be further fundamentally researched and discloses the possible molecular pathogenesis of TAPVC.
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spelling pubmed-62654812018-12-07 Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection Shi, Xin Cheng, Liangping Jiao, XianTing Chen, Bo Li, Zixiong Liang, Yulai Liu, Wei Wang, Jing Liu, Gang Xu, Yuejuan Sun, Jing Fu, Qihua Lu, Yanan Chen, Sun Front Genet Genetics Total anomalous pulmonary venous connection (TAPVC) is a rare congenital heart anomaly. Several genes have been associated TAPVC but the mechanisms remain elusive. To search novel CNVs and candidate genes, we screened a cohort of 78 TAPVC cases and 100 healthy controls for rare copy number variants (CNVs) using whole exome sequencing (WES). Then we identified pathogenic CNVs by statistical comparisons between case and control groups. After that, we identified altogether eight pathogenic CNVs of seven candidate genes (PCSK7, RRP7A, SERHL, TARP, TTN, SERHL2, and NBPF3). All these seven genes have not been described previously to be related to TAPVC. After network analysis of these candidate genes and 27 known pathogenic genes derived from the literature and publicly database, PCSK7 and TTN were the most important genes for TAPVC than other genes. Our study provides novel candidate genes potentially related to this rare congenital birth defect (CHD) which should be further fundamentally researched and discloses the possible molecular pathogenesis of TAPVC. Frontiers Media S.A. 2018-11-23 /pmc/articles/PMC6265481/ /pubmed/30532766 http://dx.doi.org/10.3389/fgene.2018.00559 Text en Copyright © 2018 Shi, Cheng, Jiao, Chen, Li, Liang, Liu, Wang, Liu, Xu, Sun, Fu, Lu and Chen. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Genetics
Shi, Xin
Cheng, Liangping
Jiao, XianTing
Chen, Bo
Li, Zixiong
Liang, Yulai
Liu, Wei
Wang, Jing
Liu, Gang
Xu, Yuejuan
Sun, Jing
Fu, Qihua
Lu, Yanan
Chen, Sun
Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection
title Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection
title_full Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection
title_fullStr Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection
title_full_unstemmed Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection
title_short Rare Copy Number Variants Identify Novel Genes in Sporadic Total Anomalous Pulmonary Vein Connection
title_sort rare copy number variants identify novel genes in sporadic total anomalous pulmonary vein connection
topic Genetics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6265481/
https://www.ncbi.nlm.nih.gov/pubmed/30532766
http://dx.doi.org/10.3389/fgene.2018.00559
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