Congenital heart disease risk loci identified by genome-wide association study in European patients.


Journal

The Journal of clinical investigation
ISSN: 1558-8238
Titre abrégé: J Clin Invest
Pays: United States
ID NLM: 7802877

Informations de publication

Date de publication:
19 01 2021
Historique:
received: 30 06 2020
accepted: 12 11 2020
pubmed: 18 11 2020
medline: 10 9 2021
entrez: 17 11 2020
Statut: ppublish

Résumé

Genetic factors undoubtedly affect the development of congenital heart disease (CHD) but still remain ill defined. We sought to identify genetic risk factors associated with CHD and to accomplish a functional analysis of SNP-carrying genes. We performed a genome-wide association study (GWAS) of 4034 White patients with CHD and 8486 healthy controls. One SNP on chromosome 5q22.2 reached genome-wide significance across all CHD phenotypes and was also indicative for septal defects. One region on chromosome 20p12.1 pointing to the MACROD2 locus identified 4 highly significant SNPs in patients with transposition of the great arteries (TGA). Three highly significant risk variants on chromosome 17q21.32 within the GOSR2 locus were detected in patients with anomalies of thoracic arteries and veins (ATAV). Genetic variants associated with ATAV are suggested to influence the expression of WNT3, and the variant rs870142 related to septal defects is proposed to influence the expression of MSX1. We analyzed the expression of all 4 genes during cardiac differentiation of human and murine induced pluripotent stem cells in vitro and by single-cell RNA-Seq analyses of developing murine and human hearts. Our data show that MACROD2, GOSR2, WNT3, and MSX1 play an essential functional role in heart development at the embryonic and newborn stages.

Identifiants

pubmed: 33201861
pii: 141837
doi: 10.1172/JCI141837
pmc: PMC7810487
doi:
pii:

Types de publication

Clinical Trial Journal Article Multicenter Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Subventions

Organisme : British Heart Foundation
ID : CH/13/2/30154
Pays : United Kingdom
Organisme : British Heart Foundation
ID : RG/15/12/31616
Pays : United Kingdom
Organisme : British Heart Foundation
ID : RG/F/21/110050
Pays : United Kingdom

Commentaires et corrections

Type : CommentIn

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Auteurs

Harald Lahm (H)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Meiwen Jia (M)

Department of Translational Research in Psychiatry, Max Planck Institute of Psychiatry Munich, Munich, Germany.

Martina Dreßen (M)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Felix Wirth (F)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Nazan Puluca (N)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Ralf Gilsbach (R)

Institute for Cardiovascular Physiology, Goethe University, Frankfurt am Main, Germany.
DZHK (German Centre for Cardiovascular Research), Partner site RheinMain, Frankfurt am Main, Germany.

Bernard D Keavney (BD)

Division of Cardiovascular Sciences, School of Medical Sciences, Faculty of Biology, Medicine and Health, The University of Manchester, Manchester, United Kingdom.
Manchester Heart Centre, Manchester University NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, United Kingdom.

Julie Cleuziou (J)

Department of Congenital and Paediatric Heart Surgery, German Heart Center Munich, Munich, Germany.

Nicole Beck (N)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Olga Bondareva (O)

Institute of Experimental and Clinical Pharmacology and Toxicology, Faculty of Medicine, University of Freiburg, Freiburg, Germany.

Elda Dzilic (E)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Melchior Burri (M)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Karl C König (KC)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Johannes A Ziegelmüller (JA)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Claudia Abou-Ajram (C)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Irina Neb (I)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Zhong Zhang (Z)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Stefanie A Doppler (SA)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.

Elisa Mastantuono (E)

Institute of Human Genetics, German Research Center for Environmental Health, Helmholtz Center Munich, Neuherberg, Germany.
Institute of Human Genetics, Klinikum rechts der Isar, Technical University of Munich, Munich, Germany.

Peter Lichtner (P)

Institute of Human Genetics, German Research Center for Environmental Health, Helmholtz Center Munich, Neuherberg, Germany.

Gertrud Eckstein (G)

Institute of Human Genetics, German Research Center for Environmental Health, Helmholtz Center Munich, Neuherberg, Germany.

Jürgen Hörer (J)

Department of Congenital and Paediatric Heart Surgery, German Heart Center Munich, Munich, Germany.

Peter Ewert (P)

Department of Pediatric Cardiology and Congenital Heart Disease, German Heart Center Munich, Munich, Germany.

James R Priest (JR)

Department of Pediatrics, Division of Pediatric Cardiology, Stanford University School of Medicine, Palo Alto, California, USA.

Lutz Hein (L)

Institute of Experimental and Clinical Pharmacology and Toxicology, Faculty of Medicine, University of Freiburg, Freiburg, Germany.
BIOSS, Center for Biological Signaling Studies, University of Freiburg, Freiburg, Germany.

Rüdiger Lange (R)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.
DZHK (German Center for Cardiovascular Research) - Partner Site Munich Heart Alliance, Munich, Germany.

Thomas Meitinger (T)

Institute of Human Genetics, German Research Center for Environmental Health, Helmholtz Center Munich, Neuherberg, Germany.
Institute of Human Genetics, Klinikum rechts der Isar, Technical University of Munich, Munich, Germany.
DZHK (German Center for Cardiovascular Research) - Partner Site Munich Heart Alliance, Munich, Germany.

Heather J Cordell (HJ)

Population Health Sciences Institute, Faculty of Medical Sciences, Newcastle University, International Centre for Life, Central Parkway, Newcastle upon Tyne, United Kingdom.

Bertram Müller-Myhsok (B)

Department of Translational Research in Psychiatry, Max Planck Institute of Psychiatry Munich, Munich, Germany.
Munich Cluster of Systems Biology, SyNergy, Munich, Germany.
Institute of Translational Medicine, University of Liverpool, Liverpool, United Kingdom.

Markus Krane (M)

Department of Cardiovascular Surgery, Division of Experimental Surgery, Institute Insure (Institute for Translational Cardiac Surgery), German Heart Center Munich, Munich, Germany.
DZHK (German Center for Cardiovascular Research) - Partner Site Munich Heart Alliance, Munich, Germany.

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