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Published in: Netherlands Heart Journal 7-8/2023

Open Access 20-07-2023 | Sudden Cardiac Death | Review Article

SCN5A-1795insD founder variant: a unique Dutch experience spanning 7 decades

Authors: Virginnio M. Proost, Maarten P. van den Berg, Carol Ann Remme, Arthur A. M. Wilde

Published in: Netherlands Heart Journal | Issue 7-8/2023

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Abstract

The SCN5A-1795insD founder variant is a unique SCN5A gene variant found in a large Dutch pedigree that first came to attention in the late 1950s. To date, this is still one of the largest and best described SCN5A founder families worldwide. It was the first time that a single pathogenic variant in SCN5A proved to be sufficient to cause a sodium channel overlap syndrome. Affected family members displayed features of Brugada syndrome, cardiac conduction disease and long QT syndrome type 3, thus encompassing features of both loss and gain of sodium channel function. This brief summary takes us past 70 years of clinical experience and over 2 decades of research. It is remarkable to what extent researchers and clinicians have managed to gain understanding of this complex phenotype in a relatively short time. Extensive clinical, genetic, electrophysiological and molecular studies have provided fundamental insights into SCN5A and the cardiac sodium channel Nav1.5.
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Metadata
Title
SCN5A-1795insD founder variant: a unique Dutch experience spanning 7 decades
Authors
Virginnio M. Proost
Maarten P. van den Berg
Carol Ann Remme
Arthur A. M. Wilde
Publication date
20-07-2023
Publisher
Bohn Stafleu van Loghum
Published in
Netherlands Heart Journal / Issue 7-8/2023
Print ISSN: 1568-5888
Electronic ISSN: 1876-6250
DOI
https://doi.org/10.1007/s12471-023-01799-8

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