Fig. 4

All mutant larvae present a motor deficit at 5 dpf and reduced survival. (a) Representative image of a 24-well plate free swim arena and movement paths (6 per genotype) of larvae aged 5 dpf. (b) Quantification of mean velocity. Specific mean swim velocities are as follows: wild type, 7.17 mm/s; chchd10^−/−, 3.72 mm/s; chchd2^−/−, 3.80 mm/s, and double chchd10^−/− and chchd2^−/−, 2.87 mm/s. All mutant zebrafish swam at significantly lower mean velocities when compared to wild type zebrafish (chchd10^−/−, p = .0002; chchd2^−/−, p = .0002; double chchd10^−/− and chchd2^−/−, p < .0001). Data represented as individual data points ± SEM. A Kruskal–Wallis test was used to assess significance followed by a Dunn's multiple comparisons test. Sample sizes (n) are noted in parentheses. Double asterisks represent p < .01. (c) Survival rates were significantly reduced in all mutants, with no double chchd10^−/− and chchd2^−/− surviving past 15 days. Significance was assessed using Mantel–Cox test, and all data points were compared to wild type survival (chchd10^−/−, p < .0001; chchd2^−/−, p = .0005; double chchd10^−/− and chchd2^−/−, p < .0001). Sample sizes (n) are noted in parentheses.