Novel G1481V and Q1491H SCN5A Mutations Linked to Long QT Syndrome Destabilize the Nav1.5 Inactivation State.


Journal

CJC open
ISSN: 2589-790X
Titre abrégé: CJC Open
Pays: United States
ID NLM: 101763635

Informations de publication

Date de publication:
Mar 2021
Historique:
received: 22 09 2020
accepted: 27 09 2020
entrez: 29 3 2021
pubmed: 30 3 2021
medline: 30 3 2021
Statut: epublish

Résumé

Na The Na The Q1491H mutant channel exhibited a lower current density, a persistent Na The 5- and 12-week-old infants displayed prolonged QT intervals. Our analyses of the Q1491H and G1481V mutations correlated with the clinical diagnosis. The observed biophysical dysfunctions associated with both mutations were most likely responsible for the sudden deaths of the 2 infants. Le canal Na Le canal Na Le canal mutant Q1491H montre une densité de courant plus faible, un courant Na Les deux bébés âgés respectivement de cinq 5 et 12 semaines montraient une prolongation des intervalles QT. Nos analyses des mutations Q1491H et G1481V montrent une corrélation avec le diagnostic clinique. Les dysfonctions biophysiques observées qui sont associées aux deux mutations étaient fort probablement responsables des morts subites des deux bébés.

Sections du résumé

BACKGROUND BACKGROUND
Na
METHODS METHODS
The Na
RESULTS RESULTS
The Q1491H mutant channel exhibited a lower current density, a persistent Na
CONCLUSIONS CONCLUSIONS
The 5- and 12-week-old infants displayed prolonged QT intervals. Our analyses of the Q1491H and G1481V mutations correlated with the clinical diagnosis. The observed biophysical dysfunctions associated with both mutations were most likely responsible for the sudden deaths of the 2 infants.
INTRODUCTION BACKGROUND
Le canal Na
MÉTHODES UNASSIGNED
Le canal Na
RÉSULTATS UNASSIGNED
Le canal mutant Q1491H montre une densité de courant plus faible, un courant Na
CONCLUSIONS CONCLUSIONS
Les deux bébés âgés respectivement de cinq 5 et 12 semaines montraient une prolongation des intervalles QT. Nos analyses des mutations Q1491H et G1481V montrent une corrélation avec le diagnostic clinique. Les dysfonctions biophysiques observées qui sont associées aux deux mutations étaient fort probablement responsables des morts subites des deux bébés.

Autres résumés

Type: Publisher (fre)
Le canal Na

Identifiants

pubmed: 33778442
doi: 10.1016/j.cjco.2020.09.023
pii: S2589-790X(20)30157-8
pmc: PMC7984979
doi:

Types de publication

Journal Article

Langues

eng

Pagination

256-266

Commentaires et corrections

Type : ErratumIn

Informations de copyright

© 2020 Canadian Cardiovascular Society. Published by Elsevier Inc.

Références

FEBS Lett. 2009 Mar 4;583(5):890-6
pubmed: 19302788
Circulation. 2018 Sep 18;138(12):1195-1205
pubmed: 29959160
Circulation. 2020 Feb 11;141(6):418-428
pubmed: 31983240
Am J Physiol. 1979 Apr;236(4):H561-7
pubmed: 434221
Genet Med. 2015 May;17(5):405-24
pubmed: 25741868
Genome Med. 2019 Dec 31;12(1):3
pubmed: 31892348
Am J Physiol. 1996 Aug;271(2 Pt 2):H498-506
pubmed: 8770089
J Gen Physiol. 1996 May;107(5):643-58
pubmed: 8740377
Am J Physiol Heart Circ Physiol. 2011 Jan;300(1):H288-99
pubmed: 21076026
J Mol Cell Cardiol. 2003 Dec;35(12):1513-21
pubmed: 14654377
Int J Legal Med. 2019 Nov;133(6):1733-1742
pubmed: 31455979
J Am Coll Cardiol. 2005 Jul 19;46(2):331-7
pubmed: 16022964
Nature. 1995 Aug 24;376(6542):683-5
pubmed: 7651517
Cell. 1995 Mar 10;80(5):805-11
pubmed: 7889574
N Engl J Med. 1998 Jun 11;338(24):1709-14
pubmed: 9624190
PLoS One. 2012;7(5):e38331
pubmed: 22675453
Cell. 2020 Jan 9;180(1):122-134.e10
pubmed: 31866066
Circ Res. 1995 Sep;77(3):584-92
pubmed: 7641328
N Engl J Med. 2000 Jul 27;343(4):262-7
pubmed: 10911008
Heart Rhythm. 2010 Jun;7(6):771-8
pubmed: 20226894
JAMA. 1995 Mar 8;273(10):783-9
pubmed: 7861572
Genome Med. 2019 Nov 29;11(1):77
pubmed: 31783775
J Gen Physiol. 2015 Feb;145(2):93-106
pubmed: 25624448
Can J Cardiol. 2011 Mar-Apr;27(2):232-45
pubmed: 21459272
J Biol Chem. 1998 Jan 9;273(2):1121-9
pubmed: 9422778
J Physiol. 2012 Oct 15;590(20):5123-39
pubmed: 22826127
Cytogenet Cell Genet. 1995;68(1-2):67-70
pubmed: 7956363
J Cardiovasc Pharmacol. 2010 Jun;55(6):539-43
pubmed: 20555231

Auteurs

Quentin Plumereau (Q)

CERVO Brain Research Center, Quebec City, Quebec, Canada.

Olivier Theriault (O)

CERVO Brain Research Center, Quebec City, Quebec, Canada.

Valérie Pouliot (V)

CERVO Brain Research Center, Quebec City, Quebec, Canada.

Adrien Moreau (A)

Inserm U1046, CNRS UMR 9214, Université de Montpellier, Montpellier, France.

Elodie Morel (E)

Lyon Reference Center for Inherited Arrhythmias, Louis Pradel Cardiovascular Hospital, Bron, France.

Véronique Fressart (V)

Centre de Génétique Moléculaire et Chromosomique, Hôpital Pitié-Salpêtrière, Paris, France.

Isabelle Denjoy (I)

Hôpital Bichat Claude Bernard, Paris, France.

Antoine Delinière (A)

Lyon Reference Center for Inherited Arrhythmias, Louis Pradel Cardiovascular Hospital, Bron, France.

Francis Bessière (F)

Lyon Reference Center for Inherited Arrhythmias, Louis Pradel Cardiovascular Hospital, Bron, France.

Philippe Chevalier (P)

Lyon Reference Center for Inherited Arrhythmias, Louis Pradel Cardiovascular Hospital, Bron, France.
Department of Rhythmology, Louis Pradel Cardiovascular Hospital, Lyon, France.
Université de Lyon, Lyon, France.

Tamer M Gamal El-Din (TM)

Department of Pharmacology, University of Washington, Seattle, Washington, USA.

Mohamed Chahine (M)

CERVO Brain Research Center, Quebec City, Quebec, Canada.
Department of Medicine, Faculty of Medicine, Université Laval, Quebec City, Quebec, Canada.

Classifications MeSH