Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathy

Hypertrophic cardiomyopathy (HCM) is often caused by pathogenic or likely pathogenic variants, of which 30–50 % involve a variant in the gene encoding cardiac myosin-binding protein-C (MYBPC3). We generated human induced pluripotent stem cell lines from five individuals from two families carrying a...

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Main Authors: Floor W. van den Dolder, Vincent A.J. Warnaar, Yeszamin L. Onderwater, Annette F. Baas, Diederik W.D. Kuster, Jolanda van der Velden
Format: Article
Language:English
Published: Elsevier 2025-08-01
Series:Stem Cell Research
Online Access:http://www.sciencedirect.com/science/article/pii/S1873506125000479
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author Floor W. van den Dolder
Vincent A.J. Warnaar
Yeszamin L. Onderwater
Annette F. Baas
Diederik W.D. Kuster
Jolanda van der Velden
author_facet Floor W. van den Dolder
Vincent A.J. Warnaar
Yeszamin L. Onderwater
Annette F. Baas
Diederik W.D. Kuster
Jolanda van der Velden
author_sort Floor W. van den Dolder
collection DOAJ
description Hypertrophic cardiomyopathy (HCM) is often caused by pathogenic or likely pathogenic variants, of which 30–50 % involve a variant in the gene encoding cardiac myosin-binding protein-C (MYBPC3). We generated human induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant: c.2373insG (n = 2) and c.2827C > T (n = 3), with highly variable disease expression. Peripheral blood mononuclear cells were reprogrammed using episomal plasmids. All cell lines express pluripotent markers, exhibit a normal karyotype, and could differentiate into derivatives of each germ layers in vitro. These cell lines can serve as disease model to investigate HCM pathogenesis.
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institution OA Journals
issn 1873-5061
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publishDate 2025-08-01
publisher Elsevier
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series Stem Cell Research
spelling doaj-art-784294334e81418889e911bd4c2997222025-08-20T02:35:36ZengElsevierStem Cell Research1873-50612025-08-018610369710.1016/j.scr.2025.103697Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathyFloor W. van den Dolder0Vincent A.J. Warnaar1Yeszamin L. Onderwater2Annette F. Baas3Diederik W.D. Kuster4Jolanda van der Velden5Physiology, Amsterdam UMC, Location Vrije Universiteit Amsterdam, De Boelelaan 1117, Amsterdam, the Netherlands; Amsterdam Cardiovascular Sciences, Heart Failure & Arrhythmias, Amsterdam, the NetherlandsPhysiology, Amsterdam UMC, Location Vrije Universiteit Amsterdam, De Boelelaan 1117, Amsterdam, the Netherlands; Amsterdam Cardiovascular Sciences, Heart Failure & Arrhythmias, Amsterdam, the NetherlandsPhysiology, Amsterdam UMC, Location Vrije Universiteit Amsterdam, De Boelelaan 1117, Amsterdam, the Netherlands; Amsterdam Cardiovascular Sciences, Heart Failure & Arrhythmias, Amsterdam, the NetherlandsDepartment of Genetics, University Medical Center Utrecht, Utrecht University, Utrecht, the NetherlandsPhysiology, Amsterdam UMC, Location Vrije Universiteit Amsterdam, De Boelelaan 1117, Amsterdam, the Netherlands; Amsterdam Cardiovascular Sciences, Heart Failure & Arrhythmias, Amsterdam, the NetherlandsPhysiology, Amsterdam UMC, Location Vrije Universiteit Amsterdam, De Boelelaan 1117, Amsterdam, the Netherlands; Amsterdam Cardiovascular Sciences, Heart Failure & Arrhythmias, Amsterdam, the Netherlands; Corresponding author.Hypertrophic cardiomyopathy (HCM) is often caused by pathogenic or likely pathogenic variants, of which 30–50 % involve a variant in the gene encoding cardiac myosin-binding protein-C (MYBPC3). We generated human induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant: c.2373insG (n = 2) and c.2827C > T (n = 3), with highly variable disease expression. Peripheral blood mononuclear cells were reprogrammed using episomal plasmids. All cell lines express pluripotent markers, exhibit a normal karyotype, and could differentiate into derivatives of each germ layers in vitro. These cell lines can serve as disease model to investigate HCM pathogenesis.http://www.sciencedirect.com/science/article/pii/S1873506125000479
spellingShingle Floor W. van den Dolder
Vincent A.J. Warnaar
Yeszamin L. Onderwater
Annette F. Baas
Diederik W.D. Kuster
Jolanda van der Velden
Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathy
Stem Cell Research
title Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathy
title_full Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathy
title_fullStr Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathy
title_full_unstemmed Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathy
title_short Generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic Dutch MYBPC3 founder variant with variable degrees of hypertrophic cardiomyopathy
title_sort generation of induced pluripotent stem cell lines from five individuals from two families carrying a pathogenic dutch mybpc3 founder variant with variable degrees of hypertrophic cardiomyopathy
url http://www.sciencedirect.com/science/article/pii/S1873506125000479
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