Human Induced Pluripotent Stem-Cell-Derived Cardiomyocytes as Models for Genetic Cardiomyopathies

Published on Sep 6, 2019in International Journal of Molecular Sciences4.18
· DOI :10.3390/ijms20184381
Andreas Brodehl9
Estimated H-index: 9
Hans Ebbinghaus1
Estimated H-index: 1
+ 4 AuthorsHendrik Milting27
Estimated H-index: 27
View in Source
In the last few decades, many pathogenic or likely pathogenic genetic mutations in over hundred different genes have been described for non-ischemic, genetic cardiomyopathies. However, the functional knowledge about most of these mutations is still limited because the generation of adequate animal models is time-consuming and challenging. Therefore, human induced pluripotent stem cells (iPSCs) carrying specific cardiomyopathy-associated mutations are a promising alternative. Since the original discovery that pluripotency can be artificially induced by the expression of different transcription factors, various patient-specific-induced pluripotent stem cell lines have been generated to model non-ischemic, genetic cardiomyopathies in vitro. In this review, we describe the genetic landscape of non-ischemic, genetic cardiomyopathies and give an overview about different human iPSC lines, which have been developed for the disease modeling of inherited cardiomyopathies. We summarize different methods and protocols for the general differentiation of human iPSCs into cardiomyocytes. In addition, we describe methods and technologies to investigate functionally human iPSC-derived cardiomyocytes. Furthermore, we summarize novel genome editing approaches for the genetic manipulation of human iPSCs. This review provides an overview about the genetic landscape of inherited cardiomyopathies with a focus on iPSC technology, which might be of interest for clinicians and basic scientists interested in genetic cardiomyopathies.
Figures & Tables
  • References (477)
  • Citations (1)
#1Nicolò Salvarani (National Research Council)H-Index: 11
#2Silvia Crasto (National Research Council)H-Index: 2
Last.Elisa Di Pasquale (National Research Council)H-Index: 12
view all 17 authors...
4 CitationsSource
#1Wei Zhou (Mayo Clinic)H-Index: 2
#2J. Martijn Bos (Mayo Clinic)H-Index: 28
Last.Michael John Ackerman (Mayo Clinic)H-Index: 102
view all 11 authors...
2 CitationsSource
#1Hua Liu (SJTU: Shanghai Jiao Tong University)H-Index: 9
#2Ying-Jia Xu (Fudan University)H-Index: 3
Last.Yi-Qing Yang (Fudan University)H-Index: 5
view all 11 authors...
3 CitationsSource
#1Anna Gaertner-Rommel (RUB: Ruhr University Bochum)H-Index: 6
#2Jens Tiesmeier (RUB: Ruhr University Bochum)H-Index: 2
Last.Hendrik Milting (RUB: Ruhr University Bochum)H-Index: 27
view all 13 authors...
1 CitationsSource
#1Jun Guo (Capital Medical University)H-Index: 1
#2Zheng Li (Capital Medical University)H-Index: 2
Last.Wei Li (Capital Medical University)H-Index: 1
view all 9 authors...
2 CitationsSource
#1Ronald NgH-Index: 1
#2Heather R. ManringH-Index: 3
Last.Stuart G. CampbellH-Index: 13
view all 22 authors...
2 CitationsSource
#1Jason D. Roberts (UWO: University of Western Ontario)H-Index: 13
#2Nathaniel P. Murphy (The Ohio State University Wexner Medical Center)H-Index: 5
Last.Peter J. Mohler (The Ohio State University Wexner Medical Center)H-Index: 32
view all 56 authors...
3 CitationsSource
#1J. SvahnH-Index: 2
#2Pascal Laforêt (French Institute of Health and Medical Research)H-Index: 45
Last.Tanya Stojkovic (University of Paris)H-Index: 41
view all 12 authors...
2 CitationsSource
#1Haodi Wu (Stanford University)H-Index: 20
#2Huaxiao Yang (Stanford University)H-Index: 10
Last.Joseph C. Wu (Stanford University)H-Index: 91
view all 13 authors...
3 CitationsSource
#1Disheet ShahH-Index: 2
#2Laura VirtanenH-Index: 1
Last.Katriina Aalto-SetäläH-Index: 21
view all 12 authors...
1 CitationsSource
Cited By1
View next paperHuman-induced pluripotent stem cells in modeling inherited cardiomyopathies