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Hydrocephalus

  • RESEARCH ARTICLE
    ZFP423 regulates early patterning and multiciliogenesis in the hindbrain choroid plexus
    Filippo Casoni, Laura Croci, Francesca Vincenti, Paola Podini, Michela Riba, Luca Massimino, Ottavio Cremona, G. Giacomo Consalez
    Development 2020 147: dev190173 doi: 10.1242/dev.190173 Published 30 November 2020

    Summary: The zinc-finger protein 423, a transcription factor and protein-protein interaction hub, is identified as a master gene in hindbrain choroid plexus patterning and multiciliogenesis.

  • RESEARCH ARTICLE
    The FOXJ1 target Cfap206 is required for sperm motility, mucociliary clearance of the airways and brain development
    Anja Beckers, Christian Adis, Karin Schuster-Gossler, Lena Tveriakhina, Tim Ott, Franziska Fuhl, Jan Hegermann, Karsten Boldt, Katrin Serth, Ev Rachev, Leonie Alten, Elisabeth Kremmer, Marius Ueffing, Martin Blum, Achim Gossler
    Development 2020 147: dev188052 doi: 10.1242/dev.188052 Published 15 June 2020

    Summary: Evolutionarily conserved CFAP206 modulates ciliary beat frequency in multiciliated cells in Xenopus and mouse, and is required for radial spoke arrangement in mouse sperm flagella.

  • RESEARCH ARTICLE
    A mutation in Ccdc39 causes neonatal hydrocephalus with abnormal motile cilia development in mice
    Zakia Abdelhamed, Shawn M. Vuong, Lauren Hill, Crystal Shula, Andrew Timms, David Beier, Kenneth Campbell, Francesco T. Mangano, Rolf W. Stottmann, June Goto
    Development 2018 145: dev154500 doi: 10.1242/dev.154500 Published 9 January 2018

    Summary: Ccdc39-deficient ependymal cells exhibit abundant short, thin cilia that are incapable of creating the rhythmic beating pattern necessary for cerebrospinal fluid flow in the perinatal mouse brain.

  • STEM CELLS AND REGENERATION
    mTORC1 signaling and primary cilia are required for brain ventricle morphogenesis
    Philippe Foerster, Marie Daclin, Shihavuddin Asm, Marion Faucourt, Alessandra Boletta, Auguste Genovesio, Nathalie Spassky
    Development 2017 144: 201-210; doi: 10.1242/dev.138271

    Highlighted article: Primary cilia regulate ventricle morphogenesis in mice by modulating the mTORC1 pathway, highlighting a new role for mTOR signalling during brain development.

  • RESEARCH ARTICLE
    Functional antagonism of voltage-gated K+ channel α-subunits in the developing brain ventricular system
    Hongyuan Shen, Elke Bocksteins, Igor Kondrychyn, Dirk Snyders, Vladimir Korzh
    Development 2016 143: 4249-4260; doi: 10.1242/dev.140467

    Summary: Voltage-gated K+ channels establish a balance between cell proliferation and barrier properties in the neuroepithelium that lines the ventricles during development of the zebrafish brain ventricular system.

  • RESEARCH ARTICLES
    Graded phenotypic response to partial and complete deficiency of a brain-specific transcript variant of the winged helix transcription factor RFX4
    Perry J. Blackshear, Joan P. Graves, Deborah J. Stumpo, Inma Cobos, John L. R. Rubenstein, Darryl C. Zeldin
    Development 2003 130: 4539-4552; doi: 10.1242/dev.00661

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