TY - JOUR
T1 - Inherited Ventricular Arrhythmia in Zebrafish
T2 - Genetic Models and Phenotyping Tools
AU - Sieliwonczyk, Ewa
AU - Matchkov, Vladimir V
AU - Vandendriessche, Bert
AU - Alaerts, Maaike
AU - Bakkers, Jeroen
AU - Loeys, Bart
AU - Schepers, Dorien
N1 - © 2021. The Author(s), under exclusive license to Springer Nature Switzerland AG.
PY - 2021/9/18
Y1 - 2021/9/18
N2 - In the last years, the field of inheritable ventricular arrhythmia disease modelling has changed significantly with a push towards the use of novel cellular cardiomyocyte based models. However, there is a growing need for new in vivo models to study the disease pathology at the tissue and organ level. Zebrafish provide an excellent opportunity for in vivo modelling of inheritable ventricular arrhythmia syndromes due to the remarkable similarity between their cardiac electrophysiology and that of humans. Additionally, many state-of-the-art methods in gene editing and electrophysiological phenotyping are available for zebrafish research. In this review, we give a comprehensive overview of the published zebrafish genetic models for primary electrical disorders and arrhythmogenic cardiomyopathy. We summarise and discuss the strengths and weaknesses of the different technical approaches for the generation of genetically modified zebrafish disease models, as well as the electrophysiological approaches in zebrafish phenotyping. By providing this detailed overview, we aim to draw attention to the potential of the zebrafish model for studying arrhythmia syndromes at the organ level and as a platform for personalised medicine and drug testing.
AB - In the last years, the field of inheritable ventricular arrhythmia disease modelling has changed significantly with a push towards the use of novel cellular cardiomyocyte based models. However, there is a growing need for new in vivo models to study the disease pathology at the tissue and organ level. Zebrafish provide an excellent opportunity for in vivo modelling of inheritable ventricular arrhythmia syndromes due to the remarkable similarity between their cardiac electrophysiology and that of humans. Additionally, many state-of-the-art methods in gene editing and electrophysiological phenotyping are available for zebrafish research. In this review, we give a comprehensive overview of the published zebrafish genetic models for primary electrical disorders and arrhythmogenic cardiomyopathy. We summarise and discuss the strengths and weaknesses of the different technical approaches for the generation of genetically modified zebrafish disease models, as well as the electrophysiological approaches in zebrafish phenotyping. By providing this detailed overview, we aim to draw attention to the potential of the zebrafish model for studying arrhythmia syndromes at the organ level and as a platform for personalised medicine and drug testing.
U2 - 10.1007/112_2021_65
DO - 10.1007/112_2021_65
M3 - Article
C2 - 34533615
SN - 0303-4240
JO - Reviews of physiology, biochemistry and pharmacology
JF - Reviews of physiology, biochemistry and pharmacology
ER -