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Mechanistically Distinct Mouse Models for CRX-Associated Retinopathy

Figure 7

Heterozygous E168d2/+, E168d2neo/+ and R90W/+ mice have graded deficits in retinal function.

A–I. Retinal function of E168d2/+, E168d2neo/+ and R90W/+ and +/− mice was assessed by electroretinography at 1 mo (A–C), 3 mo (D–F) and 6 mo (G–I). Average peak amplitude responses for dark-adapted A-waves and B-waves and light-adapted B-waves are plotted against the log of the flash intensity (Log [cd*s/m2]). Genotype*flash intensity interactions for peak amplitude (by two-way ANOVA) were significant (p<0.05) at all ages for each wave form tested. E168d2/+ mice show severe deficits in all wave responses at each age compared to responses from either WT or E168d2neo/+ mice (green vs. black and red line, respectively). Peak responses in E168d2neo/+ mice are higher than E168d2/+ (red vs. green line), but remain significantly decreased compared to WT (red vs. black line) with exceptions for 6 mo dark-adapted B-waves (H). R90W/+ and +/− mice have mostly normal retinal function (blue or orange vs. black line) but R90W/+ have subtle significant deficits in light-adapted B-waves at 6 mo (I, blue vs. black line). (Relative to WT: *p<0.05; relative to E168d2neo/+: *‘p<0.05, brackets indicate all enclosed data points are significant). Error bars: SEM.

Figure 7

doi: https://doi.org/10.1371/journal.pgen.1004111.g007