Beyond the one gene–one disease paradigm: complex genetics and pleiotropy in inheritable cardiac disorders

M Cerrone, CA Remme, R Tadros, CR Bezzina… - Circulation, 2019 - Am Heart Assoc
Inheritable cardiac disorders, which may be associated with cardiomyopathic changes, are
often associated with increased risk of sudden death in the young. Early linkage analysis …

Human induced pluripotent stem-cell-derived cardiomyocytes as models for genetic cardiomyopathies

A Brodehl, H Ebbinghaus, MA Deutsch… - International journal of …, 2019 - mdpi.com
In the last few decades, many pathogenic or likely pathogenic genetic mutations in over
hundred different genes have been described for non-ischemic, genetic cardiomyopathies …

Maturation of iPSC-derived cardiomyocytes in a heart-on-a-chip device enables modeling of dilated cardiomyopathy caused by R222Q-SCN5A mutation

M Wauchop, N Rafatian, Y Zhao, W Chen, M Gagliardi… - Biomaterials, 2023 - Elsevier
To better understand sodium channel (SCN5A)-related cardiomyopathies, we generated
ventricular cardiomyocytes from induced pluripotent stem cells obtained from a dilated …

Arrhythmic Phenotypes Are a Defining Feature of Dilated Cardiomyopathy-Associated SCN5A Variants: A Systematic Review

S Peters, BA Thompson, M Perrin, P James… - Circulation: Genomic …, 2022 - Am Heart Assoc
Background: Variants in the SCN5A gene, that encodes the cardiac sodium channel, Nav1.
5, are associated with a highly arrhythmogenic form of dilated cardiomyopathy (DCM). Our …

Biophysical properties of NaV1.5 channels from atrial-like and ventricular-like cardiomyocytes derived from human induced pluripotent stem cells

CA Chapotte-Baldacci, M Pierre, M Djemai, V Pouliot… - Scientific Reports, 2023 - nature.com
Generating atrial-like cardiomyocytes derived from human induced pluripotent stem cells
(hiPSCs) is crucial for modeling and treating atrial-related diseases, such as atrial …

Identification of SCN5a p.C335R Variant in a Large Family with Dilated Cardiomyopathy and Conduction Disease

F Sedaghat-Hamedani, S Rebs, I El-Battrawy… - International Journal of …, 2021 - mdpi.com
Introduction: Familial dilated cardiomyopathy (DCM) is clinically variable and has been
associated with mutations in more than 50 genes. Rapid improvements in DNA sequencing …

The differential impacts of equivalent gating-charge mutations in voltage-gated sodium channels

E Elhanafy, A Akbari Ahangar, R Roth… - Journal of General …, 2025 - rupress.org
Voltage-gated sodium (Nav) channels are pivotal for cellular signaling, and mutations in Nav
channels can lead to excitability disorders in cardiac, muscular, and neural tissues. A major …

Modelling sarcomeric cardiomyopathies with human cardiomyocytes derived from induced pluripotent stem cells

LR Sewanan, SG Campbell - The Journal of physiology, 2020 - Wiley Online Library
Cardiomyocytes derived from human induced pluripotent stem cells (iPSCs) provide a
unique opportunity to understand the pathophysiological effects of genetic cardiomyopathy …

NaV1.5 knockout in iPSCs: a novel approach to study NaV1.5 variants in a human cardiomyocyte environment

M Pierre, M Djemai, H Poulin, M Chahine - Scientific reports, 2021 - nature.com
Cardiomyocytes derived from patient-specific induced pluripotent stem cells (iPSC-CMs)
successfully reproduce the mechanisms of several channelopathies. However, this …

The G213D variant in Nav1.5 alters sodium current and causes an arrhythmogenic phenotype resulting in a multifocal ectopic Purkinje-related premature contraction …

K Calloe, M Geryk, K Freude, JA Treat, VA Vold… - Europace, 2022 - academic.oup.com
Abstract Aims Variants in SCN5A encoding Nav1. 5 are associated with cardiac arrhythmias.
We aimed to determine the mechanism by which c. 638G> A in SCNA5 resulting in p …