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Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant

Arrhythmogenic cardiomyopathy is an inheritable heart disease characterized by lethal heart rhythms and abnormal contractile function. Mutations in desmoplakin (DSP), a protein linking the cardiac desmosome with intermediate filaments, are associated with arrhythmogenic cardiomyopathy. Here we gener...

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Autores principales: Loiben, Alexander, Friedman, Clayton E., Chien, Wei-Ming, Stempien-Otero, April, Lin, Shin, Yang, Kai-Chun
Formato: Online Artículo Texto
Lenguaje:English
Publicado: 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9900081/
https://www.ncbi.nlm.nih.gov/pubmed/36481506
http://dx.doi.org/10.1016/j.scr.2022.102987
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author Loiben, Alexander
Friedman, Clayton E.
Chien, Wei-Ming
Stempien-Otero, April
Lin, Shin
Yang, Kai-Chun
author_facet Loiben, Alexander
Friedman, Clayton E.
Chien, Wei-Ming
Stempien-Otero, April
Lin, Shin
Yang, Kai-Chun
author_sort Loiben, Alexander
collection PubMed
description Arrhythmogenic cardiomyopathy is an inheritable heart disease characterized by lethal heart rhythms and abnormal contractile function. Mutations in desmoplakin (DSP), a protein linking the cardiac desmosome with intermediate filaments, are associated with arrhythmogenic cardiomyopathy. Here we generated a human induced pluripotent stem cell (hiPSC) line from a patient with a heterozygous protein-truncating variant in DSP (c.1386del Leu462Serfs*22). This line has a normal karyotype and expression of pluripotency markers, and can differentiate into all three germ layers. This line is well suited for in vitro mechanistic studies of mechanism of DSP protein-truncation mutations in the context of arrhythmogenic cardiomyopathy.
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spelling pubmed-99000812023-02-06 Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant Loiben, Alexander Friedman, Clayton E. Chien, Wei-Ming Stempien-Otero, April Lin, Shin Yang, Kai-Chun Stem Cell Res Article Arrhythmogenic cardiomyopathy is an inheritable heart disease characterized by lethal heart rhythms and abnormal contractile function. Mutations in desmoplakin (DSP), a protein linking the cardiac desmosome with intermediate filaments, are associated with arrhythmogenic cardiomyopathy. Here we generated a human induced pluripotent stem cell (hiPSC) line from a patient with a heterozygous protein-truncating variant in DSP (c.1386del Leu462Serfs*22). This line has a normal karyotype and expression of pluripotency markers, and can differentiate into all three germ layers. This line is well suited for in vitro mechanistic studies of mechanism of DSP protein-truncation mutations in the context of arrhythmogenic cardiomyopathy. 2023-02 2022-11-30 /pmc/articles/PMC9900081/ /pubmed/36481506 http://dx.doi.org/10.1016/j.scr.2022.102987 Text en https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) ).
spellingShingle Article
Loiben, Alexander
Friedman, Clayton E.
Chien, Wei-Ming
Stempien-Otero, April
Lin, Shin
Yang, Kai-Chun
Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant
title Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant
title_full Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant
title_fullStr Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant
title_full_unstemmed Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant
title_short Generation of human iPSC line from an arrhythmogenic cardiomyopathy patient with a DSP protein-truncating variant
title_sort generation of human ipsc line from an arrhythmogenic cardiomyopathy patient with a dsp protein-truncating variant
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9900081/
https://www.ncbi.nlm.nih.gov/pubmed/36481506
http://dx.doi.org/10.1016/j.scr.2022.102987
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