ZFIN ID: ZDB-FIG-190107-2
Blanco-Sánchez et al., 2018 - Grxcr1 Promotes Hair Bundle Development by Destabilizing the Physical Interaction between Harmonin and Sans Usher Syndrome Proteins. Cell Reports   25:1281-1291.e4 Full text @ Cell Rep.
ADDITIONAL FIGURES
PHENOTYPE:
Fish:
Observed In:
Stage: Day 5

Fig. 3

ush1c−/− and ush1ga−/− Mutants Have Dysmorphic Hair Bundles

(A and B) Phalloidin staining of the anterior macula in ush1c siblings (A) and ush1cfh293/fh293 mutant larvae (B). The red arrowhead points to splayed hair bundles.

(C) Average width of hair bundles at half their height in ush1cfh293 siblings (n = 15 larvae) and ush1cfh293/fh293 (n = 14 larvae).

(D) Number of hair bundles splayed in ush1cfh293 siblings (n = 15 larvae) and ush1cfh293/fh293 (n = 14 larvae).

(E–H) Phalloidin staining of the anterior macula at in ush1ga siblings (E and G), ush1gab1342/b1342 (F), and ush1gab1240/b1240 mutant larvae (H).

(I) Average hair bundle width at half their height in ush1gab1342 siblings (n = 10 larvae), ush1gab1342/b1342 (n = 11 larvae), ush1gab1240 siblings (n = 28 larvae), and ush1gab1240/b1240 mutant larvae (n = 9 larvae).

(J) Number of hair bundles splayed in ush1gab1342 siblings (n = 15 larvae), ush1gab1342/b1342 (n = 13 larvae), ush1gab1240 siblings (n = 35 larvae), and ush1gab1240/b1240 mutant larvae (n = 14 larvae).

Data are represented as mean ± SEM. ∗∗p < 0.01; 5 dpf.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
ush1cfh293/fh293 standard conditions Day 5 hair cell anterior macula decreased width, abnormal
Day 5 hair cell anterior macula increased height, abnormal
Day 5 hair cell anterior macula morphology, abnormal
Day 5 hair cell anterior macula splayed, abnormal
ush1gab1240/b1240 standard conditions Day 5 hair cell anterior macula decreased amount, abnormal
Day 5 hair cell anterior macula decreased width, abnormal
Day 5 hair cell anterior macula increased height, abnormal
Day 5 hair cell anterior macula morphology, abnormal
Day 5 hair cell anterior macula splayed, abnormal
ush1gab1342/b1342 standard conditions Day 5 hair cell anterior macula decreased amount, abnormal
Day 5 hair cell anterior macula decreased width, abnormal
Day 5 hair cell anterior macula increased height, abnormal
Day 5 hair cell anterior macula morphology, abnormal
Day 5 hair cell anterior macula splayed, abnormal
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 @ Cell Rep.