FIGURE

Fig. 2

ID
ZDB-FIG-250203-31
Publication
Naef et al., 2025 - Modeling sacsin depletion in Danio Rerio offers new insight on retinal defects in ARSACS
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Fig. 2

Visual impairment in the sacs-/- larval zebrafish. (A-A') Zebrafish sacs-/- mutant larvae at 120 hpf showed reduced optokinetic response. The frequency was calculated considering the number of saccades per minute for each larva examined (N = 40 for each group, sacs-/- and controls). (B-B′) Light-Dark preference test was performed according to (Rock et al., 2022). At 120 h post-fertilization, sacs-/- mutant larvae displayed freezing behavior under light conditions (measured as crossed time per minute) and showed no preference for the bright/white area during the dark phase (measured as percentage of time spent in the light area (N = 15 for each group,sacs-/-and controls). ****p < 0.0001 was calculated by Kruskal Wallis test multiple comparisons. (C-C′) Zebrafish sacs-/- mutant larvae at 120 hpf showed an affected visual motor response (VMR). Sacs-/- mutants motor activity appears significantly higher in light- off conditions compared to controls siblings and greatly reduced in light-on condition (n = 20 for each group, sacs-/- and controls). ****p < 0.0001 was calculated by two-tailed Mann-Whitney test. Error bars indicate mean ± s.e.m.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Observed In:
Stage: Day 5

Phenotype Detail
Acknowledgments
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