PUBLICATION
Using Zebrafish Animal Model to Study the Genetic Underpinning and Mechanism of Arrhythmogenic Cardiomyopathy
- Authors
- Niu, Y., Sun, Y., Liu, Y., Du, K., Xu, X., Ding, Y.
- ID
- ZDB-PUB-230226-70
- Date
- 2023
- Source
- International Journal of Molecular Sciences 24(4): (Review)
- Registered Authors
- Ding, Yonghe, Xu, Xiaolei
- Keywords
- arrhythmogenic cardiomyopathy, disease model, genetic, zebrafish
- MeSH Terms
-
- Animals
- Arrhythmias, Cardiac
- Arrhythmogenic Right Ventricular Dysplasia*/genetics
- Death, Sudden, Cardiac
- Models, Animal
- Zebrafish*
- PubMed
- 36835518 Full text @ Int. J. Mol. Sci.
Citation
Niu, Y., Sun, Y., Liu, Y., Du, K., Xu, X., Ding, Y. (2023) Using Zebrafish Animal Model to Study the Genetic Underpinning and Mechanism of Arrhythmogenic Cardiomyopathy. International Journal of Molecular Sciences. 24(4):.
Abstract
Arrhythmogenic cardiomyopathy (ACM) is largely an autosomal dominant genetic disorder manifesting fibrofatty infiltration and ventricular arrhythmia with predominantly right ventricular involvement. ACM is one of the major conditions associated with an increased risk of sudden cardiac death, most notably in young individuals and athletes. ACM has strong genetic determinants, and genetic variants in more than 25 genes have been identified to be associated with ACM, accounting for approximately 60% of ACM cases. Genetic studies of ACM in vertebrate animal models such as zebrafish (Danio rerio), which are highly amenable to large-scale genetic and drug screenings, offer unique opportunities to identify and functionally assess new genetic variants associated with ACM and to dissect the underlying molecular and cellular mechanisms at the whole-organism level. Here, we summarize key genes implicated in ACM. We discuss the use of zebrafish models, categorized according to gene manipulation approaches, such as gene knockdown, gene knock-out, transgenic overexpression, and CRISPR/Cas9-mediated knock-in, to study the genetic underpinning and mechanism of ACM. Information gained from genetic and pharmacogenomic studies in such animal models can not only increase our understanding of the pathophysiology of disease progression, but also guide disease diagnosis, prognosis, and the development of innovative therapeutic strategies.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping