PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTN
Centrosome structure, function, and number are finely regulated at the cellular level to ensure normal mammalian development. Here, we characterize PPP1R35 as a novel bona fide centrosomal protein and demonstrate that it is critical for centriole elongation. Using quantitative super-resolution micro...
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doaj-22b919373ea24439ac5040689cf6116a2021-05-05T16:07:42ZengeLife Sciences Publications LtdeLife2050-084X2018-08-01710.7554/eLife.37846PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTNAndrew Michael Sydor0https://orcid.org/0000-0003-3585-0446Etienne Coyaud1Cristina Rovelli2https://orcid.org/0000-0003-3171-6696Estelle Laurent3Helen Liu4Brian Raught5Vito Mennella6https://orcid.org/0000-0002-4842-9012Cell Biology Program, The Hospital for Sick Children, Toronto, CanadaPrincess Margaret Cancer Centre, University Health Network, Toronto, CanadaCell Biology Program, The Hospital for Sick Children, Toronto, CanadaPrincess Margaret Cancer Centre, University Health Network, Toronto, CanadaCell Biology Program, The Hospital for Sick Children, Toronto, CanadaPrincess Margaret Cancer Centre, University Health Network, Toronto, Canada; Department of Medical Biophysics, University of Toronto, Ontario, CanadaCell Biology Program, The Hospital for Sick Children, Toronto, Canada; Department of Biochemistry, University of Toronto, Ontario, CanadaCentrosome structure, function, and number are finely regulated at the cellular level to ensure normal mammalian development. Here, we characterize PPP1R35 as a novel bona fide centrosomal protein and demonstrate that it is critical for centriole elongation. Using quantitative super-resolution microscopy mapping and live-cell imaging we show that PPP1R35 is a resident centrosomal protein located in the proximal lumen above the cartwheel, a region of the centriole that has eluded detailed characterization. Loss of PPP1R35 function results in decreased centrosome number and shortened centrioles that lack centriolar distal and microtubule wall associated proteins required for centriole elongation. We further demonstrate that PPP1R35 acts downstream of, and forms a complex with, RTTN, a microcephaly protein required for distal centriole elongation. Altogether, our study identifies a novel step in the centriole elongation pathway centered on PPP1R35 and elucidates downstream partners of the microcephaly protein RTTN.https://elifesciences.org/articles/37846centrosomecentriolecentriole elongationcentriole duplicationstructured illumination microscopyprimary recessive microcephaly |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Andrew Michael Sydor Etienne Coyaud Cristina Rovelli Estelle Laurent Helen Liu Brian Raught Vito Mennella |
spellingShingle |
Andrew Michael Sydor Etienne Coyaud Cristina Rovelli Estelle Laurent Helen Liu Brian Raught Vito Mennella PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTN eLife centrosome centriole centriole elongation centriole duplication structured illumination microscopy primary recessive microcephaly |
author_facet |
Andrew Michael Sydor Etienne Coyaud Cristina Rovelli Estelle Laurent Helen Liu Brian Raught Vito Mennella |
author_sort |
Andrew Michael Sydor |
title |
PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTN |
title_short |
PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTN |
title_full |
PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTN |
title_fullStr |
PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTN |
title_full_unstemmed |
PPP1R35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein RTTN |
title_sort |
ppp1r35 is a novel centrosomal protein that regulates centriole length in concert with the microcephaly protein rttn |
publisher |
eLife Sciences Publications Ltd |
series |
eLife |
issn |
2050-084X |
publishDate |
2018-08-01 |
description |
Centrosome structure, function, and number are finely regulated at the cellular level to ensure normal mammalian development. Here, we characterize PPP1R35 as a novel bona fide centrosomal protein and demonstrate that it is critical for centriole elongation. Using quantitative super-resolution microscopy mapping and live-cell imaging we show that PPP1R35 is a resident centrosomal protein located in the proximal lumen above the cartwheel, a region of the centriole that has eluded detailed characterization. Loss of PPP1R35 function results in decreased centrosome number and shortened centrioles that lack centriolar distal and microtubule wall associated proteins required for centriole elongation. We further demonstrate that PPP1R35 acts downstream of, and forms a complex with, RTTN, a microcephaly protein required for distal centriole elongation. Altogether, our study identifies a novel step in the centriole elongation pathway centered on PPP1R35 and elucidates downstream partners of the microcephaly protein RTTN. |
topic |
centrosome centriole centriole elongation centriole duplication structured illumination microscopy primary recessive microcephaly |
url |
https://elifesciences.org/articles/37846 |
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