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Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging

The majority of cilia are formed and maintained by the highly conserved process of intraflagellar transport (IFT). Mutations in IFT genes lead to ciliary structural defects and systemic disorders termed ciliopathies. Here we show that the severely truncated sensory cilia of hypomorphic IFT mutants i...

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Autores principales: Cornils, Astrid, Maurya, Ashish K., Tereshko, Lauren, Kennedy, Julie, Brear, Andrea G., Prahlad, Veena, Blacque, Oliver E., Sengupta, Piali
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5131903/
https://www.ncbi.nlm.nih.gov/pubmed/27906968
http://dx.doi.org/10.1371/journal.pgen.1006325
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author Cornils, Astrid
Maurya, Ashish K.
Tereshko, Lauren
Kennedy, Julie
Brear, Andrea G.
Prahlad, Veena
Blacque, Oliver E.
Sengupta, Piali
author_facet Cornils, Astrid
Maurya, Ashish K.
Tereshko, Lauren
Kennedy, Julie
Brear, Andrea G.
Prahlad, Veena
Blacque, Oliver E.
Sengupta, Piali
author_sort Cornils, Astrid
collection PubMed
description The majority of cilia are formed and maintained by the highly conserved process of intraflagellar transport (IFT). Mutations in IFT genes lead to ciliary structural defects and systemic disorders termed ciliopathies. Here we show that the severely truncated sensory cilia of hypomorphic IFT mutants in C. elegans transiently elongate during a discrete period of adult aging leading to markedly improved sensory behaviors. Age-dependent restoration of cilia morphology occurs in structurally diverse cilia types and requires IFT. We demonstrate that while DAF-16/FOXO is dispensable, the age-dependent suppression of cilia phenotypes in IFT mutants requires cell-autonomous functions of the HSF1 heat shock factor and the Hsp90 chaperone. Our results describe an unexpected role of early aging and protein quality control mechanisms in suppressing ciliary phenotypes of IFT mutants, and suggest possible strategies for targeting subsets of ciliopathies.
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spelling pubmed-51319032016-12-21 Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging Cornils, Astrid Maurya, Ashish K. Tereshko, Lauren Kennedy, Julie Brear, Andrea G. Prahlad, Veena Blacque, Oliver E. Sengupta, Piali PLoS Genet Research Article The majority of cilia are formed and maintained by the highly conserved process of intraflagellar transport (IFT). Mutations in IFT genes lead to ciliary structural defects and systemic disorders termed ciliopathies. Here we show that the severely truncated sensory cilia of hypomorphic IFT mutants in C. elegans transiently elongate during a discrete period of adult aging leading to markedly improved sensory behaviors. Age-dependent restoration of cilia morphology occurs in structurally diverse cilia types and requires IFT. We demonstrate that while DAF-16/FOXO is dispensable, the age-dependent suppression of cilia phenotypes in IFT mutants requires cell-autonomous functions of the HSF1 heat shock factor and the Hsp90 chaperone. Our results describe an unexpected role of early aging and protein quality control mechanisms in suppressing ciliary phenotypes of IFT mutants, and suggest possible strategies for targeting subsets of ciliopathies. Public Library of Science 2016-12-01 /pmc/articles/PMC5131903/ /pubmed/27906968 http://dx.doi.org/10.1371/journal.pgen.1006325 Text en © 2016 Cornils et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Research Article
Cornils, Astrid
Maurya, Ashish K.
Tereshko, Lauren
Kennedy, Julie
Brear, Andrea G.
Prahlad, Veena
Blacque, Oliver E.
Sengupta, Piali
Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging
title Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging
title_full Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging
title_fullStr Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging
title_full_unstemmed Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging
title_short Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging
title_sort structural and functional recovery of sensory cilia in c. elegans ift mutants upon aging
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5131903/
https://www.ncbi.nlm.nih.gov/pubmed/27906968
http://dx.doi.org/10.1371/journal.pgen.1006325
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