FIGURE

Figure 1

ID

ZDB-FIG-220827-14

Publication

Crouzier et al., 2022 - Morphological, behavioral and cellular analyses revealed different phenotypes in Wolfram syndrome wfs1a and wfs1b zebrafish mutant lines

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Figure 1

Characterization of wfs1 mutant zebrafish lines. (A) Schematic molecular structure of Wfs1a and Wfs1b proteins with mutation. The stars represent the punctual mutations, blue for Wfs1a mutation and orange for Wfs1b. (B) Relative wfs1a and wfs1b mRNA levels assessed by qPCR in wfs1a^C825X and wfs1b^W493X zebrafish larvae, normalized against zef1α reference gene. (C) Schematic representation of the different measurements of the larva. (D) Representative images of wild-type and homozygous mutant wfs1 larvae. Images to the right show the ear in the wild-type (upper image) and wfs1a^C825X mutant (the lower image). (E) Measurement of body size, (F) eye diameter, (G) ear area in lateral view, (H) anterior and (I) posterior otoliths area. Scale bars, 200 μm in (D), 10 μm in the insets. Error bars represent mean ± SEM and the number of fish is indicated below the columns. ^**P < 0.01, ^***P < 0.0001; unpaired t-test.

Expression Data

Genes:	wfs1a wfs1b
Fish:	AB wfs1a^{sa11465/sa11465} (AB) wfs1b^{sa16422/sa16422} (AB)
Anatomical Term:	whole organism
Stage:	Day 5

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	wfs1a^{sa11465/sa11465} (AB) wfs1b^{sa16422/sa16422} (AB)
Observed In:	eye otic vesicle otolith whole organism
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 @ Hum. Mol. Genet.