FIGURE

Fig. 3.

ID

ZDB-FIG-241125-23

Publication

Noble et al., 2024 - Shared and unique consequences of Joubert Syndrome gene dysfunction on the zebrafish central nervous system

Other Figures

All Figure Page

Back to All Figure Page

Fig. 3.

Loss of primary cilia in cerebellar neurons of cc2d2a and talpid3 mutants. (A) Representative whole-mount maximum projection confocal immunofluorescence images showing parallel fibres of granule cells (anti-Vglut1 – cyan), Purkinje cells (anti-Pvalb7 – magenta) and olig2+ eurydendroid cells [Tg(olig2:EGFP) transgenic line – green] in the cerebellum of wild-type zebrafish larvae at 5 dpf. Images show a dorsal view with anterior to the left. (B-G) Whole-mount maximum projection confocal immunofluorescence images showing primary cilia labelled with the Tg(ubi:Arl13b-mCherry) transgenic line (Arl13b – yellow) and basal bodies (BB – green) labelled using anti-γ-Tubulin (C,E-G) or using the Tg(β-actin:GFP-centrin) transgenic line (B,D). Purkinje cells (PC – magenta) are marked using anti-Pvalb7 (B-D) and eurydendroid cells (EC – green) are marked by the Tg(olig2:EGFP) transgenic line (E-G). Note the strong decrease in cilia numbers in both mutants (cc2d2a^−/− in C and F; talpid3^−/− in D and G) compared to control (B and E). Insets show a magnified view of one representative cilium [from top to bottom: composite image (B3-G3), Arl13b (B4-G4) and BB (B5-G5)]. Images show a dorsal view of 5 dpf larvae with anterior to the left. Scale bars: 10 µm. (H-I) Violin plots showing a statistically significant reduction in Arl13b+ cilia within the mutant Purkinje cell layer (H) and olig2+ eurydendroid cell layer (I) compared to controls. Each data point represents one larva, and violin plot represents median (thick line) and quartiles (dashed line). ****P≤0.0001. Welch's ANOVA with post-hoc Dunnett's T3 multiple comparisons test. For Purkinje cell analysis, control n=25 (N=5), cc2d2a^−/−n=10 (N=3), talpid3^−/−n=15 (N=2) larvae. For eurydendroid cell analysis, control n=23 (N=4), cc2d2a^−/−n=8 (N=2), talpid3^−/−n=16 (N=2) larvae. N denotes the number of independent experiments (i.e. larvae from independent clutches) used for analysis. Analyses were carried out using mz cc2d2a^−/− and zygotic talpid3^−/− mutants.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

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 @ Biol. Open