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Fig. 5 Adult chchd10P83L/P83L zebrafish recover expression of Chchd10 and do not display a significant motor deficit in either a free swim or swim tunnel assay. A, Example images of adult 6-month-old wild type and chchd10P83L/P83L zebrafish. Scale bar represents 1 cm. Bi, Representative immunoblot showing expression of Chchd10 and Chchd2 in whole brain lysate. Bii, Quantification of Chchd10 and Chchd2 levels relative to Sdhb. No differences were found in the expression levels of Chchd10 and Chchd2 between wild type and chchd10P83L/P83L zebrafish brains: Chchd10, (t (4) = 0.02, p = 0.98); Chchd2, (t (4) = 0.67, p = 0.54). Ci, Adult 6-month-old zebrafish were placed into an open field arena and mean swim velocity was measured. No differences in mean free swim velocity were found across all the genotypes (F (3,39) = 1.24, p > 0.05). Mean free swim velocity and sample sizes are as follows: wild type (4.28 cm/s, n = 10); chchd10?/? (5.49 cm/s, n = 11); chchd2?/? (5.18 cm/s, n = 12); chchd10P83L/P83L (6.03 cm/s, n = 12). Cii, Free swim velocity was re-assessed at 16 months of age. No significant differences in mean swim velocity were observed in our fish models though there was a trend of chchd10P83L/P83L zebrafish swimming with a slower mean velocity compared to wild type (Kruskal-Wallis test, H (4) = 7.21, p = 0.07). Measures of mean velocity and sample sizes were as follows: wild type (6.21 cm/s, n = 12); chchd10?/? (5.92 cm/s, n = 11); chchd2?/? (5.97 cm/s, n = 12); and chchd10P83L/P83L (4.21 cm/s, n = 10). Di, Adult zebrafish were placed into a swim tunnel and a sprint swim assay was performed to calculate Umax. A single significant difference was observed of Umax (F (3,33) = 3.73, p = 0.02) between chchd10?/? and chchd2 ?/? (p = 0.027) at 6 months of age. Mean Umax and sample sizes were as follows: wild type (76.58 cm/s, n = 10); chchd10?/? (88.69 cm/s, n = 7); chchd2?/? (61.76 cm/s, n = 10); and chchd10P83L/P83L (64.26 cm/s, n = 10). Dii, The sprint swim assay was re-examined at 14 months of age and while a non-parametric ANOVA yield that the data contained significant differences among genotypes (H (4) = 8.76, p = 0.03) a Kruskal-Wallis post-hoc multiple comparisons test was unable to find where the differences lay (p > 0.05). Mean Umax and sample sizes were as follows: wild type (43.81 cm/s, n = 14); chchd10?/? (51.80 cm/s, n = 12); chchd2 ?/? (40.68 cm/s, n = 11); and chchd10 P83L/P83L (48.43 cm/s, n = 10). Data is represented as individual measures with mean ± S.E.M.