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

9 Citations (Scopus)

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

Fingerprint

Cilia
proteins
Fibers
Basal Bodies
Axoneme
entry
Proteins
fibers
Membrane Proteins
assembly
biological evolution
Membranes
Hydrolethalus syndrome
depletion
anomalies
membranes
C elegans HYLS-1 protein

ASJC Scopus subject areas

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

Cite this

The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate. / Wei, Qing; Zhang, Yingyi; Schouteden, Clementine; Zhang, Yuxia; Zhang, Qing; Dong, Jinhong; Wonesch, Veronika; Ling, Kun; Dammermann, Alexander; Hu, Jinghua.

In: Nature Communications, Vol. 7, 12437, 18.08.2016.

Research output: Contribution to journalArticle

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, vol. 7, 12437. https://doi.org/10.1038/ncomms12437
Wei, Qing ; Zhang, Yingyi ; Schouteden, Clementine ; Zhang, Yuxia ; Zhang, Qing ; Dong, Jinhong ; Wonesch, Veronika ; Ling, Kun ; Dammermann, Alexander ; Hu, Jinghua. / The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate. In: Nature Communications. 2016 ; Vol. 7.
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AU - Dong, Jinhong

AU - Wonesch, Veronika

AU - Ling, Kun

AU - Dammermann, Alexander

AU - Hu, Jinghua

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