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An essential role for DYF-11/MIP-T3 in assembling functional intraflagellar transport complexes
Södertörn University, School of Life Sciences.
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2008 (English)In: PLoS Genetics, ISSN 1553-7390, E-ISSN 1553-7404, Vol. 4, no 3, e1000044- p.Article in journal (Refereed) Published
Abstract [en]

MIP-T3 is a human protein found previously to associate with microtubules and the kinesin-interacting neuronal protein DISC1 ( Disrupted-in-Schizophrenia 1), but whose cellular function(s) remains unknown. Here we demonstrate that the C. elegans MIP-T3 ortholog DYF-11 is an intraflagellar transport (IFT) protein that plays a critical role in assembling functional kinesin motor-IFT particle complexes. We have cloned a loss of function dyf-11 mutant in which several key components of the IFT machinery, including Kinesin-II, as well as IFT subcomplex A and B proteins, fail to enter ciliary axonemes and/or mislocalize, resulting in compromised ciliary structures and sensory functions, and abnormal lipid accumulation. Analyses in different mutant backgrounds further suggest that DYF-11 functions as a novel component of IFT subcomplex B. Consistent with an evolutionarily conserved cilia-associated role, mammalian MIP-T3 localizes to basal bodies and cilia, and zebrafish mipt3 functions synergistically with the Bardet-Biedl syndrome protein Bbs4 to ensure proper gastrulation, a key cilium- and basal body-dependent developmental process. Our findings therefore implicate MIP-T3 in a previously unknown but critical role in cilium biogenesis and further highlight the emerging role of this organelle in vertebrate development.

Place, publisher, year, edition, pages
2008. Vol. 4, no 3, e1000044- p.
National Category
Genetics
Identifiers
URN: urn:nbn:se:sh:diva-14165DOI: 10.1371/journal.pgen.1000044ISI: 000255407300004Scopus ID: 2-s2.0-41949085600OAI: oai:DiVA.org:sh-14165DiVA: diva2:466985
Available from: 2011-12-18 Created: 2011-12-16 Last updated: 2014-02-26Bibliographically approved

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Publisher's full textScopushttp://www.plosgenetics.org/article/info%3Adoi%2F10.1371%2Fjournal.pgen.1000044

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CiteExportLink to record
Permanent link

Direct link
Cite
Citation style
  • apa
  • harvard1
  • ieee
  • modern-language-association-8th-edition
  • vancouver
  • Other style
More styles
Language
  • de-DE
  • en-GB
  • en-US
  • fi-FI
  • nn-NO
  • nn-NB
  • sv-SE
  • Other locale
More languages
Output format
  • html
  • text
  • asciidoc
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