title: Failure of centrosome migration causes a loss of motile cilia in talpid(3) mutants.
abstract: BACKGROUND
  Loss of function mutations in the centrosomal protein TALPID3 (KIAA0586) cause a failure of primary cilia formation in animal models and are associated with defective Hedgehog signalling. It is unclear, however, if TALPID3 is required only for primary cilia formation or if it is essential for all ciliogenesis, including that of motile cilia in multiciliate cells.
  
  
  RESULTS
  FOXJ1, a key regulator of multiciliate cell fate, is expressed in the dorsal neuroectoderm of the chicken forebrain and hindbrain at stage 20HH, in areas that will give rise to choroid plexuses in both wt and talpid(3) embryos. Wt ependymal cells of the prosencephalic choroid plexuses subsequently transition from exhibiting single short cilia to multiple long motile cilia at 29HH (E8). Primary cilia and long motile cilia were only rarely observed on talpid(3) ependymal cells. Electron microscopy determined that talpid(3) ependymal cells do develop multiple centrosomes in accordance with FOXJ1 expression, but these fail to migrate to the apical surface of ependymal cells although axoneme formation was sometimes observed.
  
  
  CONCLUSIONS
  TALPID3, which normally localises to the proximal centrosome, is essential for centrosomal migration prior to ciliogenesis but is not directly required for de novo centriologenesis, multiciliated fate, or axoneme formation.
authors.affiliation: Division of Developmental Biology, The Roslin Institute and R(D)SVS, University of Edinburgh, Easter Bush, Midlothian, UK.
authors.forename: Louise A
authors.initials: LA
authors.lastname: Stephen
authors.forename: Gemma M
authors.initials: GM
authors.lastname: Davis
authors.forename: Katie E
authors.initials: KE
authors.lastname: McTeir
authors.forename: John
authors.initials: J
authors.lastname: James
authors.forename: Lynn
authors.initials: L
authors.lastname: McTeir
authors.forename: Martin
authors.initials: M
authors.lastname: Kierans
authors.forename: Andrew
authors.initials: A
authors.lastname: Bain
authors.forename: Megan G
authors.initials: MG
authors.lastname: Davey
mesh_terms: D000818:Animals; D064652:Basal Bodies / Q000378:metabolism; D018385:Centrosome / Q000378:metabolism*; D002642:Chick Embryo; D002645:Chickens; D002923:Cilia / Q000378:metabolism*; D004027:Diencephalon / Q000378:metabolism; D051858:Forkhead Transcription Factors / Q000235:genetics / Q000378:metabolism; D017403:In Situ Hybridization; D016548:Prosencephalon / Q000378:metabolism; D013687:Telencephalon / Q000378:metabolism
chemical_list: D051858:Forkhead Transcription Factors
keywords: FOXJ1; TALPID3; basal body; centriologenesis; choroid plexus; ciliogenesis; diencephalon; distal appendage; prosencephalon; telencephalon; transition fibre