CEP41 interacts with TTLL6 and is required for localizing TTLL6 to the cilium. (a) Morpholino knockdown of zebrafish ttll6 associates with ciliary phenotypes such as curved tail (arrows), abnormal number/orientation of ear otolith (boxes), cystic kidney (arrowheads) and peripheral cardiac edema (asterisks) at different dosages indicated and show A-tubule specific defects in the outer doublet microtubules. The numbers of defective cilia were counted in both WT embryos and ttll6 morphants (n = 3 embryos, >20 cilia per animal), quantified below. (b) GFP-CEP41 was immunoprecipitated with anti-Flag antibody recognizing Flag-tagged TTLL6 from whole-cell extract (WCE), compared with GFP-empty vector. In the reciprocal co-IP experiment with anti-GFP antibody, the interaction between CEP41 and TTLL6 was confirmed. (c) Disturbed localization of TTLL6 to the cilium following Cep41 siRNA co-transfection with GFP-TTLL6 into IMCD3 cells and immunostained with either GT335 or ARL13B antibody. Arrows: cilia; Arrowheads: basal bodies. Cells expressing ciliary localized TTLL6 were counted only in siRNA-transfected cells quantified in graph. Scale bar 5 μm. *P< 0.01, **P < 0.001. Error bars = s.e.m.