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Failure of centrosome migration causes a loss of motile cilia in talpid3 mutants

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Bibliographic Details
Journal Title: Developmental Dynamics
Authors and Corporations: Stephen, Louise A., Davis, Gemma M., Mcteir, Katie E., James, John, Mcteir, Lynn, Kierans, Martin, Bain, Andrew, Davey, Megan G.
In: Developmental Dynamics, 242, 2013, 8, p. 923-931
Type of Resource: E-Article
Language: English
Summary: <jats:p><jats:styled-content>Background:</jats:styled-content> 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. <jats:styled-content>Results:</jats:styled-content> <jats:italic>FOXJ1</jats:italic>, 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 <jats:italic>wt</jats:italic> and <jats:italic>talpid<jats:sup>3</jats:sup></jats:italic> embryos. <jats:italic>Wt</jats:italic> 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 <jats:italic>talpid<jats:sup>3</jats:sup></jats:italic> ependymal cells. Electron microscopy determined that <jats:italic>talpid<jats:sup>3</jats:sup></jats:italic> ependymal cells do develop multiple centrosomes in accordance with <jats:italic>FOXJ1</jats:italic> expression, but these fail to migrate to the apical surface of ependymal cells although axoneme formation was sometimes observed. <jats:styled-content>Conclusions:</jats:styled-content> 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. <jats:italic>Developmental Dynamics 242:923–931, 2013</jats:italic>. © 2013 Wiley Periodicals, Inc.</jats:p>
Physical Description: 923-931
ISSN: 1058-8388
DOI: 10.1002/dvdy.23980