The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate

Qing Wei, Yingyi Zhang, Clementine Schouteden, Yuxia Zhang, Qing Zhang, Jinhong Dong, Veronika Wonesch, Kun Ling, Alexander Dammermann, Jinghua Hu

Research output: Contribution to journalArticlepeer-review

14 Scopus citations


Transition fibres (TFs), together with the transition zone (TZ), are basal ciliary structures thought to be crucial for cilium biogenesis and function by acting as a ciliary gate to regulate selective protein entry and exit. Here we demonstrate that the centriolar and basal body protein HYLS-1, the C. Elegans orthologue of hydrolethalus syndrome protein 1, is required for TF formation, TZ organization and ciliary gating. Loss of HYLS-1 compromises the docking and entry of intraflagellar transport (IFT) particles, ciliary gating for both membrane and soluble proteins, and axoneme assembly. Additional depletion of the TF component DYF-19 in hyls-1 mutants further exacerbates TZ anomalies and completely abrogates ciliogenesis. Our data support an important role for HYLS-1 and TFs in establishment of the ciliary gate and underline the importance of selective protein entry for cilia assembly.

Original languageEnglish (US)
Article number12437
JournalNature communications
StatePublished - Aug 18 2016

ASJC Scopus subject areas

  • Physics and Astronomy(all)
  • Chemistry(all)
  • Biochemistry, Genetics and Molecular Biology(all)


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