Fig. 8

Several markers of the mitochondrial stress response are upregulated in our double chchd10^−/− and chchd2^−/− model at 5 dpf. (a) All mitochondrial integrated stress response (mt-ISR) upstream activators were significantly increased in the double chchd10^−/− and chchd2^−/− model including atf4a (p = .019); atf4b (p = .01); atf5a (p = .0001); atf5b (p = .0014); chop (p = .0029). (b) Several downstream targets are upregulated in the double chchd10^−/− and chchd2^−/− model including htra2 (p = .0002); fgf21 (p = .0062), but not clpp (p = .1799). (c) Oxygen responsive transcript cox4i2a was significantly increased in our double chchd10^−/− and chchd2^−/− model (p = .017). Data is represented as mean ± SEM. Sample sizes (n) are noted in parentheses. Significant differences from wild type larvae are represented by either a single asterisk (p < .05) or double asterisk (p < .01) using a one-way ANOVA followed by a Tuckey's multiple comparison test.