Cargando…
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...
Autores principales: | , , , , , |
---|---|
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 |
_version_ | 1784882767763341312 |
---|---|
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. |
format | Online Article Text |
id | pubmed-9900081 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT loibenalexander generationofhumanipsclinefromanarrhythmogeniccardiomyopathypatientwithadspproteintruncatingvariant AT friedmanclaytone generationofhumanipsclinefromanarrhythmogeniccardiomyopathypatientwithadspproteintruncatingvariant AT chienweiming generationofhumanipsclinefromanarrhythmogeniccardiomyopathypatientwithadspproteintruncatingvariant AT stempienoteroapril generationofhumanipsclinefromanarrhythmogeniccardiomyopathypatientwithadspproteintruncatingvariant AT linshin generationofhumanipsclinefromanarrhythmogeniccardiomyopathypatientwithadspproteintruncatingvariant AT yangkaichun generationofhumanipsclinefromanarrhythmogeniccardiomyopathypatientwithadspproteintruncatingvariant |