Fig. 3.

usp8 knockout zebrafish displays ciliopathy-related phenotypes. a Wild-type (WT) and usp8-knockout (usp8-KO) zebrafish with cystic kidney (arrows), hydrocephalus (arrowhead), and microphthalmia (white arrows) at 5 dpf. Scale bar, 500 μm. b Incidence of cystic kidney in WT (n = 75) and usp8 KO (n = 101) at 4 or 5 dpf. c–e Immunofluorescence analysis of cilia of pronephric duct of WT and usp8-KO at 27 hpf (c, sagittal plane) and at 4 dpf (d, transverse plane). Green, acetylated-tubulin; red, actin; blue: nuclei. Scale bars, 10 μm. Pronephric duct lumen diameter of WT (n = 11) and usp8 KO (n = 7) at 4 dpf are shown as mean ± SD in e. **p < 0.01, two-tailed unpaired student’s t-tests