ZFIN ID: ZDB-FIG-220827-14
Crouzier et al., 2022 - Morphological, behavioral and cellular analyses revealed different phenotypes in Wolfram syndrome wfs1a and wfs1b zebrafish mutant lines. Human molecular genetics   31(16):2711-2727 Full text @ Hum. Mol. Genet.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Genes:
Fish:
Anatomical Term:
Stage: Day 5
PHENOTYPE:
Fish:
Observed In:
Stage: Day 5

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 wfs1aC825X and wfs1bW493X 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 wfs1aC825X 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.

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
wfs1a AB standard conditions Day 5 whole organism RTPCR
wfs1asa11465/sa11465 (AB) standard conditions Day 5 whole organism RTPCR
wfs1bsa16422/sa16422 (AB) standard conditions Day 5 whole organism RTPCR
wfs1b AB standard conditions Day 5 whole organism RTPCR
wfs1asa11465/sa11465 (AB) standard conditions Day 5 whole organism RTPCR
wfs1bsa16422/sa16422 (AB) standard conditions Day 5 whole organism RTPCR
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
wfs1asa11465/sa11465 (AB) standard conditions Day 5 eye normal diameter, normal
Day 5 otic vesicle increased area, abnormal
Day 5 otolith normal area, normal
Day 5 whole organism normal length, normal
wfs1bsa16422/sa16422 (AB) standard conditions Day 5 eye normal diameter, normal
Day 5 otic vesicle normal area, normal
Day 5 otolith normal area, normal
Day 5 whole organism wfs1a expression decreased amount, abnormal
Day 5 whole organism wfs1b expression decreased amount, abnormal
Day 5 whole organism normal length, normal
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.