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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 () and Jinghua Hu ()
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Qing Wei: Mayo Clinic
Yingyi Zhang: Mayo Clinic
Clementine Schouteden: Max F. Perutz Laboratories, Vienna Biocenter (VBC), University of Vienna
Yuxia Zhang: Mayo Clinic
Qing Zhang: Mayo Clinic
Jinhong Dong: Mayo Clinic
Veronika Wonesch: Max F. Perutz Laboratories, Vienna Biocenter (VBC), University of Vienna
Kun Ling: Mayo Clinic
Alexander Dammermann: Max F. Perutz Laboratories, Vienna Biocenter (VBC), University of Vienna
Jinghua Hu: Mayo Clinic

Nature Communications, 2016, vol. 7, issue 1, 1-10

Abstract: 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.

Date: 2016
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Persistent link: https://EconPapers.repec.org/RePEc:nat:natcom:v:7:y:2016:i:1:d:10.1038_ncomms12437

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DOI: 10.1038/ncomms12437

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