Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Review
. 2018 Oct:138:116-125.
doi: 10.1016/j.pbiomolbio.2018.05.013. Epub 2018 May 30.

Phenotyping cardiomyopathy in adult zebrafish

Alexey V Dvornikov  1 , Pieter P de Tombe  2 , Xiaolei Xu  3
Affiliations
Review

Phenotyping cardiomyopathy in adult zebrafish

Alexey V Dvornikov et al. Prog Biophys Mol Biol. 2018 Oct.

Abstract

Hypertrophic cardiomyopathy (HCM) is usually manifested by increased myofilament Ca2+ sensitivity, excessive contractility, and impaired relaxation. In contrast, dilated cardiomyopathy (DCM) originates from insufficient sarcomere contractility and reduced cardiac pump function, subsequently resulting in heart failure. The zebrafish has emerged as a new model of human cardiomyopathy with high-throughput screening, which will facilitate the discovery of novel genetic factors and the development of new therapies. Given the small hearts of zebrafish, better phenotyping tools are needed to discern different types of cardiomyopathy, such as HCM and DCM. This article reviews the existing models of cardiomyopathy, available morphologic and functional methods, and current understanding of the different types of cardiomyopathy in adult zebrafish.

Keywords: Cardiac remodeling; Cardiomyopathy; Sarcomere; Zebrafish.

PubMed Disclaimer

Figures

Figure 1
Figure 1
Assessment of sarcomere function in zebrafish heart. A, A single myofibril attached to glass microtools. B, Traces from the preliminary experiment showing increased relaxation time tLIN in mybpc3−/ − mutant fish (in red).
See this image and copyright information in PMC

References

    1. Ahmad F, Seidman JG, Seidman CE. The genetic basis for cardiac remodeling. Annu Rev Genomics Hum Genet. 2005;6:185–216. - PubMed
    1. Alves ML, Dias FAL, Gaffin RD, Simon JN, Montminy EM, Biesiadecki BJ, Hinken AC, Warren CM, Utter MS, Davis RTR, Sakthivel S, Robbins J, Wieczorek DF, Solaro RJ, Wolska BM. Desensitization of myofilaments to Ca2+ as a therapeutic target for hypertrophic cardiomyopathy with mutations in thin filament proteins. Circ Cardiovasc Genet. 2014;7:132–143. - PMC - PubMed
    1. Asimaki A, Kapoor S, Plovie E, Karin Arndt A, Adams E, Liu Z, James CA, Judge DP, Calkins H, Churko J, Wu JC, MacRae CA, Kleber AG, Saffitz JE. Identification of a new modulator of the intercalated disc in a zebrafish model of arrhythmogenic cardiomyopathy. Sci Transl Med. 2014;6:240ra74. - PMC - PubMed
    1. Barefield D, Kumar M, Gorham J, Seidman JG, Seidman CE, de Tombe PP, Sadayappan S. Haploinsufficiency of MYBPC3 exacerbates the development of hypertrophic cardiomyopathy in heterozygous mice. J Mol Cell Cardiol. 2015;79:234–43. - PMC - PubMed
    1. Belus A, Piroddi N, Scellini B, Tesi C, D’Amati G, Girolami F, Yacoub M, Cecchi F, Olivotto I, Poggesi C. The familial hypertrophic cardiomyopathy-associated myosin mutation R403Q accelerates tension generation and relaxation of human cardiac myofibrils. J Physiol. 2008;586:3639–44. - PMC - PubMed

Publication types