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. 2019 Nov 14;4(22):e130516. doi: 10.1172/jci.insight.130516

Figure 5. Rescue of the ift88 morphant phenotype with cosuppression of usp38 in 5dpf zebrafish larvae.

Figure 5

(A) Mislocalization of rhodopsin staining (green) is trapped in the cytoplasm of the outer nuclear layer (ONL) cells in ift88 morphant (arrowheads), but localization is restored to normal in double bbs4/usp38 mutants. Blue, DAPI. Top and bottom panels of images were taken at 20×, and midpanel of images were taken at 40×. (B) The rhodopsin located in the cytoplasmic region (indicated by dashed red line in A) of ONL cells was quantified by ImageJ (NIH). Suppression of ift88 causes a significant increase of rhodopsin mislocalization. Cosuppression of usp38 rescues this defect (1-way ANOVA). **P < 0.01; ****P < 0.0001. (C) Assessment of visual function by visual startle response (VSR). Knockdown of ift88 leads to deficient startle response upon light stimulation. However, the visual ability can be improved by cosuppression of usp38 (1-way ANOVA). ****P < 0.0001.