Fig. 1

Loss of function of Pontin leads to cilia-associated phenotypes in zebrafish. (A) A wild-type (WT) sibling and a pontin^{hi1055B/hi1055B} (hi1055B) mutant at 3 days post fertilization (dpf). Boxed regions are enlarged in the lower middle and right panels. The mutant shows ventral body curvature and kidney cysts (arrow). (B) Transverse sections of the glomerular neck region of a wild-type sibling and a pontin^{hi1055B/hi1055B} mutant at 50 h post fertilization (hpf). Arrow points to the enlarged lumen in the mutant. (C) Absence of pontin (pon) transcripts in pontin^{hi1055B/hi1055B} mutants. RT-PCR on cDNA from 5 dpf wild-type siblings and pontin^{hi1055B/hi1055B} mutants using primers targeting regions on the 5′ or 3′ side of the proviral insertion. actin is used as a loading control. 40 embryos were pooled per sample. (D) Representative images of embryos at 3 dpf from hi1055B/+ incrosses injected with eGFP or pontin-eGFP mRNA. (E) Significantly reduced frequency of ventral body curvature and kidney cyst in pontin-eGFP mRNA-injected hi1055B/+ incross embryos, as compared with eGFP mRNA-injected control groups. Data are represented as average±s.d. from three independent experiments. **P<0.01.