Applications of the zebrafish model for drug discovery and muscle disease research. Schematic highlighting multiple uses for the zebrafish model, including human disease modeling, repurposing of existing clinically approved drugs, discovery of novel drug compounds, toxicology, phenotype-based high-throughput screening, and characterization of the relationship between the structure of a molecule and its (therapeutic) activity.

Standard zebrafish drug treatment design. The timeline shows a standard zebrafish drug treatment workflow, as described in many of the studies listed in Table 1. Below the timeline are several variables and parameters that need to be considered when conducting drug screening and small-molecule testing in zebrafish. Adult fish of the genetic background of choice are bred, and at 1 day post fertilization (dpf), embryos are treated with an appropriate dose of the drug (or control compound). Media containing the drug are changed each day, and the typical duration of exposure lasts 3 days. At 4 dpf, muscle integrity is examined via a number of available assays, as described in this Special Article. Notably, the duration of drug treatment and outcomes examined can be altered depending on the specific disease/model of interest.