Motor function and muscle pathology in cdkl5−/− fish. (A-C) Quantification of normalized distance travelled (A), speed (B) and time active (C) of cdkl5+/+, cdkl5+/− and cdkl5−/− fish at 6 dpf. Data are mean±s.e.m. for three independent experiments (n=38, 21 and 15 for cdkl5+/+; n=17, 39 and 19 for cdkl5+/−; and n=27, 25 and 14 for cdkl5−/− fish per experiment). *P<0.05; ****P<0.0001; NS, not significant (one-way ANOVA). (D) Maximum intensity projection of a confocal image series of α-myosin and α-Actinin2 antibody staining of cdkl5+/+ and cdkl5−/− fish at 2 dpf. (E) Maximum intensity projections of confocal images of α-myosin antibody staining of cdkl5+/+ and cdkl5−/− fish at 6 dpf. Scale bars: 100 µm (D); 10 µm (E).
|