Zebrafish muscular disease models towards drug discovery

Abstract

Background: Zebrafish is an amenable vertebrate model useful for the study of development and genetics. Small molecule screenings in zebrafish have successfully identified several drugs that affect developmental process. Objective: This review covers the basics of zebrafish muscle system such as muscle development and muscle defects. It also reviews the potential use of zebrafish for chemical screening with regards to muscle disorders. Conclusion: During embryogenesis, zebrafish start to coil their body by contracting trunk muscles 17 h postfertilization, indicating that a motor circuit and skeletal muscle are functionally developed at early stages. Mutagenesis screens in zebrafish have identified many motility mutants that display morphological or functional defects in the CNS, clustering defects of acetylcholine receptors at the neuromuscular junctions or pathological defects of muscles. Most of the muscular mutants are useful as animal models of human muscle disease such as muscle dystrophy. As zebrafish live in water, pharmacological drugs are easily assayable during development, and thus zebrafish may be used to determine novel drugs that mitigate muscle disease.

Keywords::

• development
• locomotion
• muscle
• mutant
• zebrafish