Mechanistically Distinct Mouse Models for CRX-Associated Retinopathy
Figure 9
Graded changes in CRX target gene expression in heterozygous E168d2/+, E168d2neo/+ and R90W/+ mice.
A–D. Paraffin embedded sagittal retinal sections of 1 mo WT and the indicated heterozygous mutant mice were stained with mouse monoclonal anti-Rhodopsin RetP-1 antibody (Chemicon) (RHO, red) and DAPI nuclear conterstaining (blue), and imaged by widefield fluorescence at 40×. E168d2/+ shows reduced rod OS length and mislocalized RHO in ONL. E–H. qRT-PCR analysis of four CRX target genes, Rho, Arr3, Opn1mw, Opn1sw in the indicated heterozygous mice at P10 and P21 (*p<0.05; bracketed *FDR p<0.09; Error bars: SEM). Note at P10, the expression of Opn1mw and Opn1sw CRX target genes are reduced in all mutant models. However, at P21, expression recovers in R90W/+ and +/− mice, while remaining reduced in E168d2/+ and E168d2neo/+ mice.