16-P002 The developmental mutant talpid3 lacks primary cilia

نویسندگان

  • Fiona Bangs
  • Yili Yin
  • Robert Paton
  • Alan Prescott
  • John James
  • Megan Davey
  • Paul Whitley
  • Grigory Genikhovich
  • Ulrich Technau
  • James Briscoe
  • Dave Burt
  • Cheryll Tickle
چکیده

Marc A. Willaredt, Kerstin Hasenpusch-Theil, Humphrey Gardner, Igor Kitanovic, Vera Hirschfeld-Warneken, Christian Gojak, Karin Gorgas, C. Lulu Bradford, Joachim Spatz, Stefan Woelfl, Thomas Theil, Kerry L. Tucker 1 Interdisciplinary Center for Neurosciences, University of Heidelberg, Heidelberg, Germany 2 Dept. of Anatomy, University of Heidelberg, Heidelberg, Germany 3 Centres for Neuroscience Research and Integrative Physiology, University of Edinburgh, Edinburgh, United Kingdom 4 Novartis Institutes forBioMedicalResearch, Cambridge, MA, United States 5 Institute of Pharmacy and Molecular Biotechnology, University of Heidelberg, Heidelberg, Germany 6 Institute for Physical Chemistry, Dept. of Biophysical Chemistry, University of Heidelberg, Heidelberg, Germany 7 Max Planck Institute for Metals Research, Stuttgart, Germany

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منابع مشابه

The Talpid3 gene (KIAA0586) encodes a centrosomal protein that is essential for primary cilia formation.

The chicken talpid(3) mutant, with polydactyly and defects in other embryonic regions that depend on hedgehog (Hh) signalling (e.g. the neural tube), has a mutation in KIAA0568. Similar phenotypes are seen in mice and in human syndromes with mutations in genes that encode centrosomal or intraflagella transport proteins. Such mutations lead to defects in primary cilia, sites where Hh signalling ...

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Generation of mice with functional inactivation of talpid3, a gene first identified in chicken.

Specification of digit number and identity is central to digit pattern in vertebrate limbs. The classical talpid(3) chicken mutant has many unpatterned digits together with defects in other regions, depending on hedgehog (Hh) signalling, and exhibits embryonic lethality. The talpid(3) chicken has a mutation in KIAA0586, which encodes a centrosomal protein required for the formation of primary c...

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Targeted mutation of the talpid3 gene in zebrafish reveals its conserved requirement for ciliogenesis and Hedgehog signalling across the vertebrates.

Using zinc-finger nuclease-mediated mutagenesis, we have generated mutant alleles of the zebrafish orthologue of the chicken talpid3 (ta3) gene, which encodes a centrosomal protein that is essential for ciliogenesis. Animals homozygous for these mutant alleles complete embryogenesis normally, but manifest a cystic kidney phenotype during the early larval stages and die within a month of hatchin...

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The CP110-interacting proteins Talpid3 and Cep290 play overlapping and distinct roles in cilia assembly

We have identified Talpid3/KIAA0586 as a component of a CP110-containing protein complex important for centrosome and cilia function. Talpid3 assembles a ring-like structure at the extreme distal end of centrioles. Ablation of Talpid3 resulted in an aberrant distribution of centriolar satellites involved in protein trafficking to centrosomes as well as cilia assembly defects, reminiscent of los...

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Talpid3-Binding Centrosomal Protein Cep120 Is Required for Centriole Duplication and Proliferation of Cerebellar Granule Neuron Progenitors

Granule neuron progenitors (GNPs) are the most abundant neuronal type in the cerebellum. GNP proliferation and thus cerebellar development require Sonic hedgehog (Shh) secreted from Purkinje cells. Shh signaling occurs in primary cilia originating from the mother centriole. Centrioles replicate only once during a typical cell cycle and are responsible for mitotic spindle assembly and organizati...

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عنوان ژورنال:
  • Mechanisms of Development

دوره 126  شماره 

صفحات  -

تاریخ انتشار 2009