Fig. 4: Longitudinal analysis of retinal structure and function in Gpatch11Δ5/Δ5 mice.
From: GPATCH11 variants cause mis-splicing and early-onset retinal dystrophy with neurological impairment
a Cone- and rod-specific ERG responses to light in wild-type (WT, black) and Gpatch11Δ5/Δ5 (red) mice at post-natal days 15, 21, 30, 60, 90, 120, 150, and 180. Significance of the difference in a-wave and B-wave amplitudes between age-matched mutant Gpatch11Δ5/Δ5 and wild-type mice was determined through a post hoc Sidak test following a two-way ANOVA. Bars show means ± SEM from nine mice per genotype. n.s. not significant. *p ≤ 0.05, **p ≤ 0.01, ***p ≤ 0.001, ****p ≤ 0.0001. b Spider graph presenting outer nuclear layer (ONL) thickness in wild-type (WT, black) and Gpatch11Δ5/Δ5 (red) mice at 15, 60, 120, and 180 days. The ONL thickness of age-matched mutant Gpatch11Δ5/Δ5 and wild-type mice were compared using a two-way ANOVA with a post hoc Sidak test. Bars represent means ± SEM from three mice per genotype. *p ≤ 0.05, **p ≤ 0.01, ***p ≤ 0.001, ****p ≤ 0.0001. c Immunostaining of retina sections with anti-GPATCH11 (magenta) antibody. Nuclei are stained with DAPI (blue). Scale bars, 50 µm. d Quantification of GPATCH11 protein expression in the ONL, inner nuclear layer (INL), and ganglion cell layer (GCL) in wild-type (WT, black) and mutant Gpatch11Δ5/Δ5 (red) mice. The mean intensity of GPATCH11 of age-matched mutant Gpatch11Δ5/Δ5 and wild-type mice was compared using a two-way ANOVA with a post-hoc Sidak test. Bars represent means ± SEM from three biological replicates. n.s. not significant. *p ≤ 0.05, **p ≤ 0.01, ***p ≤ 0.001. Source data are provided as a Source Data file.
