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 journalArticle

12 Scopus citations

Abstract

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
Volume7
DOIs
StatePublished - Aug 18 2016

ASJC Scopus subject areas

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

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    Wei, Q., Zhang, Y., Schouteden, C., Zhang, Y., Zhang, Q., Dong, J., Wonesch, V., Ling, K., Dammermann, A., & Hu, J. (2016). The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate. Nature communications, 7, [12437]. https://doi.org/10.1038/ncomms12437