FIGURE

Fig. 5

ID

ZDB-FIG-220630-40

Publication

Reurink et al., 2022 - Scrutinizing pathogenicity of the USH2A c.2276 G > T; p.(Cys759Phe) variant

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Fig. 5

Electroretinogram recordings show that ush2a^{p.(Cys771Phe)} mutants are vision impaired.

a Representative electroretinograms of a p.(Cys771Phe) zebrafish and a wildtype sibling at 5 days post-fertilization. b The maximum B wave amplitude was significantly lower in p.(Cys771Phe) zebrafish as compared to wildtype siblings (unpaired t test). The average wildtype amplitude was normalized to 1. Each datapoint corresponds to recordings from an individual larvae (mean ± SEM). *p: 0.0445. c A comparative analysis of ush2a^rmc1 knock-out larvae and age- and strain-matched wildtype larvae was shown to result in a similar and significant decrease in maximum B wave amplitude. Again, the average wildtype amplitude was normalized to 1. Each datapoint corresponds to recordings from an individual larvae (unpaired t test, mean ± SEM). *p: 0.0345.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	ush2a^rmc1/rmc1 (TL) ush2a^{rmc016Tg/rmc016Tg} (TL)
Observed In:	visual perception
Stage:	Day 5

Phenotype Detail

Acknowledgments

This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ NPJ Genom Med