Fig. 2

Larval chchd10^−/−, chchd2^−/−, and double chchd10^−/− and chchd2^−/− display morphological defects and motor impairment. (a) Representative images of 56 hpf larvae. (b) Quantification of yolk sacs defects. Larval chchd10⁻/⁻ showing reduced yolk sack size compared to wild type larvae (p < .0001). (c) Quantification of heart defects. Both chchd2^−/− (p < .0001) and double chchd10⁻/⁻ and chchd2⁻/⁻ (p < .0001) larvae display cardiac abnormalities. (d) Representative traces of touch-evoked motor response 2 dpf larvae (n = 10 per genotype). Quantification of swim distance with wild type larvae swimming for significantly longer distances than chchd10^−/− (p = .0016) and double chchd10^−/− and chchd2^−/− (p = .0089) larvae (e). Quantification of maximum swim velocity revealed that wild type larvae swam at a maximum velocity that was significantly faster than double chchd10^−/− and chchd2^−/− (p = .0063) larvae (f). No differences were found when examining mean swim velocity across our genetic groupings (g). Data shown as individual data points ± SEM. Significance was assessed using Kruskal–Wallis test followed by Dunn's post hoc test for swimming distance or by one-way ANOVA and Tuckey's multiple comparison test for maximum velocity and mean distance measures. Significant differences from wild type larvae are represented by either a single asterisk (p < .05) or double asterisk (p < .01), and samples sizes (n) are in parentheses.