Fig. 6
From: Evaluating the association of biallelic OGDHL variants with significant phenotypic heterogeneity
Zebrafish ogdhl F0 mutant displays motor neuron abnormalities. A–L Representative image of the trunk region of uninjected, Cas9-injected, ogdhl F0 and F0 + OGDHL in Tg(olig2:DsRed2;mnx1:GFP);Albino embryos at 3 dpf. M The measurement of axon length was started from spinal cord to the end of latest posterior branch (indicated by red arrow) of caudal primary motor neurons (CaP) (indicated by red line). Axon lengths were calculated as a percentage difference compared to the mean value of uninjected embryos. Each dot represents one axon. Uninjected = 24, Cas9-injected = 24, ogdhl F0 = 30, and F0 + OGDHL = 26. N The measurements of axon angle (indicated by magenta lines, start from spinal cord). Each dot represents one axon. n = 30 axons for each group. Error bars = mean ± SD. The mean value of each group was indicated at the bottom of the respective bar in the figure. Statistical significance in M and N was calculated by Brown–Forsthye and Welch’s ANOVA with Dunnett’s T3 multiple comparisons test: not significant (ns) p ≥ 0.05, **p < 0.01, and ****p < 0.0001. O Cartoon figures indicate aberrant motor axons observed in ogdhl F0 embryos such as cross somite segment (blue hash) or missing EGFP signal (magenta asterisk). P Quantification of aberrant motor axon as indicated in O. n = 30 axons for each group