TY - JOUR
T1 - The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate
AU - Wei, Qing
AU - Zhang, Yingyi
AU - Schouteden, Clementine
AU - Zhang, Yuxia
AU - Zhang, Qing
AU - Dong, Jinhong
AU - Wonesch, Veronika
AU - Ling, Kun
AU - Dammermann, Alexander
AU - Hu, Jinghua
N1 - Publisher Copyright:
© The Author(s) 2016.
PY - 2016/8/18
Y1 - 2016/8/18
N2 - 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.
AB - 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.
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U2 - 10.1038/ncomms12437
DO - 10.1038/ncomms12437
M3 - Article
C2 - 27534274
AN - SCOPUS:84983268306
SN - 2041-1723
VL - 7
JO - Nature communications
JF - Nature communications
M1 - 12437
ER -