FIGURE

FIGURE 2

ID

ZDB-FIG-250411-2

Publication

Strachan et al., 2025 - Novel in vivo models of autosomal optic atrophy reveal conserved pathological changes in neuronal mitochondrial structure and function

Other Figures

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FIGURE 2

Loss of Opa1 impairs visual function in zebrafish. (A) Schematic of OKR assays which were used to assess visual function in <131 hpf larvae. No gross morphology differences are distinguished between opa1 crispants and buffer injected controls. (B–D) Visual acuity responses of <131 hpf opa1 crispants and controls to drums with 0.2 (B) and 0.02 (C) cycles per degree (cpd) and 0.2 cpd with reduced (20%) black/ white contrast (D). (E) A representative PCR genotyping electrophoresis gel from opa1^+/+, opa1^+/− and opa1^−/− larvae. opa1 mutation produces a 932 bp deletion band. No gross morphological differences were observed between opa1^−/− animals and opa1^+/+ siblings. (F) qPCR shows a significant reduction of opa1 transcript (normalized to B‐Actin) in both opa1^−/− and opa1^+/− animals. (G, H) Visual acuity responses of <131 hpf opa1^+/+, opa1^+/− and opa1^−/− larvae to drums with 0.2 (G) and 0.02 (H) cycles per degree. All graphs represent mean ± standard deviation (SD) saccades per minute. Statistical analysis consists of one‐way ANOVA and Tukey's multiple comparisons tests.

Expression Data

Gene:	opa1
Fish:	WT opa1^ucd16/ucd16 opa1^ucd16/+
Anatomical Term:	whole organism
Stage:	Day 4

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	opa1^ucd16/ucd16 WT + CRISPR5-opa1 + CRISPR6-opa1 opa1^ucd16/+
Knockdown Reagents:	CRISPR5-opa1 CRISPR6-opa1
Observed In:	optokinetic behavior whole organism
Stage:	Day 4

Phenotype Detail

Acknowledgments

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