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

Astrid Cornils; Ashish K Maurya; Lauren Tereshko; Julie Kennedy; Andrea G Brear; Veena Prahlad; Oliver E Blacque; Piali Sengupta

doi:10.1371/journal.pgen.1006325

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

Journal article

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

Astrid Cornils, Ashish K Maurya, Lauren Tereshko, Julie Kennedy, Andrea G Brear, Veena Prahlad, Oliver E Blacque and Piali Sengupta

PLoS genetics, Vol.12(12), pp.e1006325-e1006325

12/2016

DOI: 10.1371/journal.pgen.1006325

PMCID: PMC5131903

PMID: 27906968

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Abstract

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.

Ciliopathies - therapy

Forkhead Transcription Factors - biosynthesis

HSP90 Heat-Shock Proteins - biosynthesis

Caenorhabditis elegans - growth & development

Caenorhabditis elegans - genetics

Humans

Molecular Chaperones - genetics

Transcription Factors - biosynthesis

Transcription Factors - genetics

Forkhead Transcription Factors - genetics

Cilia - metabolism

Cilia - genetics

Ciliopathies - genetics

Microtubules - metabolism

Animals

Aging - genetics

Microtubules - genetics

HSP90 Heat-Shock Proteins - genetics

Sensory Receptor Cells - metabolism

Mutation

Caenorhabditis elegans Proteins - genetics

Caenorhabditis elegans Proteins - biosynthesis

Details

Title: Subtitle: Structural and Functional Recovery of Sensory Cilia in C. elegans IFT Mutants upon Aging
Creators: Astrid Cornils - Department of Biology and National Center for Behavioral Genomics, Brandeis University, Waltham, Massachusetts
Ashish K Maurya - Department of Biology and National Center for Behavioral Genomics, Brandeis University, Waltham, Massachusetts
Lauren Tereshko - Department of Biology and National Center for Behavioral Genomics, Brandeis University, Waltham, Massachusetts
Julie Kennedy - School of Biomolecular and Biomedical Science, University College Dublin, Belfield, Dublin, Ireland
Andrea G Brear - Department of Biology and National Center for Behavioral Genomics, Brandeis University, Waltham, Massachusetts
Veena Prahlad - Department of Biology, Aging Mind and Brain Initiative, University of Iowa, Iowa City, Iowa
Oliver E Blacque - School of Biomolecular and Biomedical Science, University College Dublin, Belfield, Dublin, Ireland
Piali Sengupta - Department of Biology and National Center for Behavioral Genomics, Brandeis University, Waltham, Massachusetts
Resource Type: Journal article
Publication Details: PLoS genetics, Vol.12(12), pp.e1006325-e1006325
DOI: 10.1371/journal.pgen.1006325
PMID: 27906968
PMCID: PMC5131903
NLM abbreviation: PLoS Genet
ISSN: 1553-7390
eISSN: 1553-7404
Publisher: United States
Grant note: R37 GM056223 / NIGMS NIH HHS\nT32 MH019929 / NIMH NIH HHS
Language: English
Date published: 12/2016
Academic Unit: Iowa Neuroscience Institute; Biology
Record Identifier: 9983991964502771

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