Fig. 1

raf is a novel zebrafish model of inherited blindness. (A) Gross morphology of raf mutant larvae and sibling controls at 131 hpf. Mutants lack an inflated swim bladder but have otherwise normal gross morphology. Scale bar = 2 mm. (B) Visual behavior was assessed by different OKR drums; standard, 0.2 cpd (cycles per degree) and 20% black contrast (bc) in < 131 hpf larvae. (C) Visual behavior was assessed by sequential exposure to colored standard OKR drums. N = 3 independent biological replicates with n = 8 larvae per replicate. Error bars indicate the mean ± SD. The midline of the error bars represents the group average. A non-parametric Mann–Whitney U-test was performed where ****p < .0001. (D) Analysis of visual function measured by electroretinogram. The response of the outer retina to a short (500 ms, rectangular black bar) flash of light at 5 increasing intensities of light (0 log = 2.8 × 103 μW/cm2) was recorded at the corneal surface of 6 dpf zebrafish larvae. n = 5 (sibling), 6 (raf mutants).