Figure 1.

Exoc5 mutant zebrafish show ciliopathy phenotypes and up-regulation of the Hippo pathway. A and B, chromatograms of Sanger sequencing reactions of wild-type (WT) and homozygous exoc5 mutant (exoc5 Mut) zebrafish. C–F, lateral view of representative WT (C and E) and exoc5 homozygous mutant (D and F) zebrafish at 3.5 dpf. exoc5 mutants showed cilia defects, which included the following: *, hydrocephaly; **, smaller eyes; ***, pericardial edema and tail curvature. Scale bars, 0.268 mm (C and D) and 0.1 mm (E and F). G, Western blot analysis of WT and exoc5 mutant zebrafish larvae. Exoc5 mutants showed loss of Exoc5 and Exoc4 proteins and up-regulation of active phosphorylated mob1 (pMOB1) protein, at 3.5 dpf. Protein lysate was isolated from n = 3 zebrafish larvae, and all the protein lysate was loaded onto each gel lane. H, quantification of phosphorylated Mob1 protein in WT versus exoc5 mutants (KO). p = 0.023. I, injection of wild-type human EXOC5 mRNA rescues the exoc5 mutant phenotype in zebrafish. This experiment was repeated twice, and a total of 120 rescued embryos were studied.