Fig. 1.

Fig. 1.

Drug screen in a zebrafish model of NEB-related nemaline myopathy. Schematic depicting the experimental flow of an unbiased drug screen in neb mutant zebrafish using the Microsource Discovery library. Heterozygous neb zebrafish (neb+/−) were mated, and neb mutant (neb−/−) offspring identified by phenotype. Once identified at 2 dpf, single neb−/− embryos were placed into individual wells of a 96-well plate, each containing a single drug at a dose of 10 μm. Embryos were incubated for 16 h, and then analyzed for movement using the Viewpoint Zebrabox. All drugs were tested in duplicate. Positive hits, as defined using a Z analysis of the movement data, were re-tested using n=16 neb (−/−) zebrafish and multiple doses of the drug. While eight drugs were initially identified as positive hits, no single compound gave a positive result upon retesting.