FIGURE 3

FIGURE 3

Nonvisual phenotypes in Opa1 deficiency models. (A) Graph represents mean ± standard deviation (SD) number of spontaneous tail coiling movements in 30 s in 24 hpf larvae. No significance (ns) detected by Krustal‐Wallis test. (B) Schematic illustrating the apparatus used for a touch response assay. Graph represents the number of 125 hpf larvae that swim the indicated distance from the starting point in response to a gentle tap on the tail. (C) Schematic illustrating the periods used for activity recording in VMR assays. Black and yellow bars indicate dark and light conditions, respectively. (D–I) Results of VMR assays involving opa1 loss of function zebrafish. No significance was detected by Krustal Wallis test. Activity traces showing activity over the course of an entire VMR assay (100 min) in <131 hpf opa1 crispant (D) and opa1^−/− (G) larva compared to relevant controls. Average ON peak activity for both on peaks in opa1 crispant (E) and opa1^−/− (H) larvae and controls. Average OFF peak activity for both peak periods in opa1 crispant (F) and opa1^−/− (I) larvae and controls. (J) Stills from videos of progeny of elav‐GAL4.UAS‐Cas9 crossed to either w¹¹¹⁸ (control) or Opa1 sgRNA (Opa1 neuronal KO) flies. (K) Graph represents % survival of Opa1 neuronal KO and control flies. n = 179–247 flies per genotype. p‐value < .0001 determined by Log‐rank (Mantel‐Cox) test.