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TitleDefining the layers of a sensory cilium with STORM and cryoelectron nanoscopy.
Journal, issue, pagesProc Natl Acad Sci U S A, Vol. 116, Issue 47, Page 23562-23572, Year 2019
Publish dateNov 19, 2019
AuthorsMichael A Robichaux / Valencia L Potter / Zhixian Zhang / Feng He / Jun Liu / Michael F Schmid / Theodore G Wensel /
PubMed AbstractPrimary cilia carry out numerous signaling and sensory functions, and defects in them, "ciliopathies," cause a range of symptoms, including blindness. Understanding of their nanometer-scale ciliary ...Primary cilia carry out numerous signaling and sensory functions, and defects in them, "ciliopathies," cause a range of symptoms, including blindness. Understanding of their nanometer-scale ciliary substructures and their disruptions in ciliopathies has been hindered by limitations of conventional microscopic techniques. We have combined cryoelectron tomography, enhanced by subtomogram averaging, with superresolution stochastic optical reconstruction microscopy (STORM) to define subdomains within the light-sensing rod sensory cilium of mouse retinas and reveal previously unknown substructures formed by resident proteins. Domains are demarcated by structural features such as the axoneme and its connections to the ciliary membrane, and are correlated with molecular markers of subcompartments, including the lumen and walls of the axoneme, the membrane glycocalyx, and the intervening cytoplasm. Within this framework, we report spatial distributions of key proteins in wild-type (WT) mice and the effects on them of genetic deficiencies in 3 models of Bardet-Biedl syndrome.
External linksProc Natl Acad Sci U S A / PubMed:31690665 / PubMed Central
MethodsEM (tomography)
Structure data

EMDB-8992:
Cryo-electron tomogram of mouse rod connecting cilium and mother centriole with C9 sub-tomogram averaging
Method: EM (tomography)

Source
  • Mus musculus (house mouse)

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