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Stem Cell Res ; 76: 103341, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38382214

ABSTRACT

Loss-of-function mutations in the PKP2 gene are associated with arrhythmogenic right ventricular cardiomyopathy (ARVC), a rare cardiac disease associated with a poor prognosis. The search for therapeutics and a better understanding of the molecular mechanisms of the disease require the development of cellular modelling. Using CRISPR/Cas9, we generated a hiPSC line with heterozygous 7-bp deletion in exon 10 of PKP2 (p.H695VfsX5). We demonstrated that hiPSCs were fully pluripotent and showed a high rate of differentiation into cardiomyocytes (iPS-CM). We also showed that PKP2 protein was expressed at the plasma membrane, with an overall decreased expression in iPS-CM indicating haploinsufficiency.


Subject(s)
Frameshift Mutation , Induced Pluripotent Stem Cells , Humans , Induced Pluripotent Stem Cells/metabolism , CRISPR-Cas Systems/genetics , Mutation , Exons/genetics , Plakophilins/genetics , Plakophilins/metabolism
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