Figure 1: DYF-19 is a functional component of TFs.

(a) Schematic of Y43F8C.4 alleles. Jhu455 and jhu546 were retrieved from a genome-wide mutagenesis screen for ciliogenesis mutants. Jhu455 and jhu546 exhibit identical mutant phenotypes in all experiments we conducted. Hereafter, the jhu455 allele with an R294>Stop mutation is the reference allele used in this report. (b) In C. elegans, the head amphids and tail phasmids are ciliated neurons and primary sensory organs. The amphid cilia consist of a bundle of ten cilia and the phasmid has two cilia. (c) Fluorescence micrographs of cilia labelled with the GFP-tagged IFT-B component OSM-6 in Wt, dyf-19(jhu455) and dyf-19 rescue strain. dyf-19 shows truncated cilia and abnormal accumulation of OSM-6::GFP at the tips of residual cilia. Arrows and arrowheads indicate the base and tip of cilia, respectively. Stars note the accumulation of OSM-6::GFP. (d) The Dyf in dyf-19(jhu455) is fully rescued by introducing a wt copy of the Y43F8C.4 gene. Data are represented as mean of five independent experiments (n=300) and error bars indicate s.d. Significant differences were identified by the Student’s t-test. *P<0.001. (e) mCherry-tagged DYF-19 specifically localizes at the ciliary base. (f) Spatial relationships between DYF-19 and TZ markers (MKS-5 and NPHP-1) and between DYF-19 and a periciliary membrane compartment (PCMC) marker (RPI-2). (g) Cartoon illustrating the TF localization of DYF-19 at the ciliary base. (h) Transmission electron microscopy reveals that dyf-19 mutants possess normal TFs. Scale bars, 200 nm in h and 5 μm in other micrographs.