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Vision recovery in late-onset RP model mice by viral gene delivery of anti-Prox1 antibody. (a) The distribution of Prox1 in the retinas of mice with heterozygous and homozygous tvrm64 mutation of Rp1 gene (Rp1m) was investigated by immunostaining. MG cells among Prox1-positive cells were determined by co-staining of Prox1 and Sox2. Boxed areas in top row are magnified in the next three rows. Sox2-positive MG nuclei are outlined by dotted-lines. (b) The thicknesses of the ONL and INL of the retinal sections were measured and relative thickness of the ONL compared to the INL of the same retinal sections is presented in the graph. **, p < 0.01; ****, p < 0.001 (One-sided Student’s t-test). (c) Relative Prox1 immunofluorescent intensity in MG in the corresponding retina, normalized to Prox1 intensity in BCs within the same image, is shown. Each dot represents the median intensity collected from one retina (data from 3 independent litters). **, p < 0.01; ***, p < 0.005; ****, p < 0.001 (One-sided Student’s t-test). (d) Rp1m/m mice were intravitreally injected with AAV2-Ctrl Ab or AAV2-αProx1 at P60, and then with EdU every 2 days for one month before sample collection to label the cells born around the injection time points. (e) The identities of these EdU-labeled cells in mouse retinas were investigated by co-immunostaining for cell type-specific markers. Immunostaining images are provided in Supplementary Fig. 19f, except for the rPR marker Rhodopsin. Boxed areas in top row are magnified in the next rows. Arrowheads, EdU-labeled cells containing Rhodopsin. (f) Number of cells expressing corresponding markers (data collected from 2 independent litters). (g) Visual acuities of the mice infected with AAV at 2 months of age (2 M) and measured at the indicated postnatal months. Error bars in the graphs represent SEM (3 independent litters). (h) Visual acuities of the mice infected with AAV at 6 M and measured at the indicated postnatal months. Error bars in the graphs represent SEM (3 independent litters).
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