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

被引:5
|
作者
Loiben, Alexander [1 ,2 ,3 ]
Friedman, Clayton E. [1 ,2 ,3 ]
Chien, Wei-Ming [1 ,2 ,3 ]
Stempien-Otero, April [1 ,2 ,3 ]
Lin, Shin [1 ,2 ,3 ]
Yang, Kai-Chun [1 ,2 ,3 ,4 ]
机构
[1] Univ Washington, Dept Med Cardiol, Seattle, WA 98109 USA
[2] Univ Washington, Ctr Cardiovasc Biol, Seattle, WA 98109 USA
[3] Univ Washington, Sch Med, Inst Stem Cell & Regenerat Med, Seattle, WA 98109 USA
[4] VA Puget Sound HCS, Cardiol Hosp Specialty Med, 1660 S Columbian Way,S111 Cardio, Seattle, WA 98108 USA
关键词
D O I
10.1016/j.scr.2022.102987
中图分类号
Q813 [细胞工程];
学科分类号
摘要
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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