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Figure 7

Expressing Gjd2b in Purkinje neurons (PNs) alone is sufficient to rescue dendritic growth deficits.

(A) Representative image of a zebrafish PN expressing cytoplasmic GFP (green) and Gjd2b tagged with mCherry (magenta) at 7 days post fertilization (dpf); scale bar is 5 µm. (B) Total dendritic branch length (TDBL) of wild type (WT), gjd2b-/-, gjd2b-rescue, and gjd2bΔ5-21 rescue (pore dead variant) PNs at 7 dpf. TDBL of gjd2b-rescue neurons is significantly increased from that of mutant PNs. TDBL of gjd2bΔ5-21 rescue PNs is not significantly different from that of gjd2b-/- PNs. (C) Change in the lengths of WT PN dendritic branches with and without Gjd2b-mCherry expression. Elongation of branches with Gjd2b is significantly more than branches without (p<0.05). Retraction of branches is similar with and without Gjd2b-mCherry puncta (Mann–Whitney U test; N = 7 neurons). (D) Copy number of CaMKII mRNA in WT and gjd2b-/- larvae. The normalized copy number of CaMKII in the mutant group is significantly higher than the WT group (p<0.05). (E) TDBL of WT, untreated gjd2b-/-, 1 µM KN-93 treated, 1 µM KN-92 treated, and 1% DMSO-treated gjd2b-/- PNs at 7 dpf. TDBL of only KN-93-treated gjd2b-/- PNs are rescued to WT levels (WT vs. KN-93, p=0.23), whereas all other groups are significantly different and lower than WT (Kruskal–Wallis, post-hoc comparison with Mann–Whitney test). Ns (number of neurons sampled) are indicated in parentheses in (B) and (D). See also Figure 7—figure supplement 1 and Supplementary file 2. Data used for quantitative analyses are available in Figure 7—source data 1.

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