Figure 4

Figure 4 Rescreen identifies 59 compounds restoring locomotion defects in the gan^del/del model

The 103 Hits identified in the gan MO‐injected fish were further tested in the gan CRISPR zebrafish line at 5 dpf. Due to the great imbalance toward positive results (all 103 compounds are Hits), the statistical analysis used a normalization of Percent Activation (%). Readout signals were calibrated and normalized to a 0–100% effect scale, in which 0% represents no effect and 100% corresponds to the locomotion activity of WT fish (See details in Appendix Table S1). Here, the cutoff for the restoration of motility was assigned to > 50% and identifies 59 Hits. Data information: For each drug, circles represent individual larvae (n = 4) and red lines show the medians for the Normalized Percent Activation.