Fig. 2

Loss of FSD1 blocks ciliogenesis at the stage of transition zone assembly. a A table summarizing the centrosome localization of the tested proteins in FSD1-depleted RPE-1 cells. CV ciliary vesicle. b–e RPE-1 cells were treated with control or FSD1 siRNAs followed by serum starvation for 48 h, then subjected to staining with indicated antibodies. Scale bars, 1 μm. b The Cep164 localization was not affected by FSD1 depletion. c FSD1 depletion did not affect the disruption of CP110 at the mother centriole. d, e Loss of FSD1 affected the localization of TMEM67 and NPHP8. f Depletion of FSD1 caused ciliary transition zone assembly defects. Representative electron micrographs of basal bodies in RPE-1 cells transfected with control (left) or FSD1 (right) siRNA, followed by serum starvation. Arrows indicate small membrane vesicles connected to FSD1-deficient centrioles through the distal appendage. Schematic diagrams summarizing the phenotype are shown. Scale bars, 200 nm. Data are presented as mean ± s.d. of three independent experiments. n number of cells. In all panels, statistical comparisons between two groups were carried out by two-tailed t-test. NS not significant, ***P < 0.001