The dynein regulatory complex is required for ciliary motility and otolith biogenesis in the inner ear

Jessica R. Colantonio, Julien Vermot, David Wu, Adam D. Langenbacher, Scott Fraser, Jau-Nian Chen and Kent L. Hill ()
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Jessica R. Colantonio: Immunology and Molecular Genetics
Julien Vermot: Biological Imaging Center, Beckman Institute, California Institute of Technology, Pasadena, California 91125, USA
David Wu: Biological Imaging Center, Beckman Institute, California Institute of Technology, Pasadena, California 91125, USA
Adam D. Langenbacher: Cell, and Developmental Biology
Scott Fraser: Biological Imaging Center, Beckman Institute, California Institute of Technology, Pasadena, California 91125, USA
Jau-Nian Chen: Cell, and Developmental Biology
Kent L. Hill: Immunology and Molecular Genetics

Nature, 2009, vol. 457, issue 7226, 205-209

Abstract: Cilia strike a balance In zebrafish — as in humans and other vertebrates — hearing and balance are mediated by mechanical sensors in the inner ear. These sensors consist of biomineralized composite crystals, called otoliths. Colantonio et al. use in vivo video microscopy of zebrafish embryos to show that fluid flow generated by cilia influences the number, growth and localization of otoliths, as well as their mineralization during development. Gene-knockdown with 'morpholino' antisense oligonucleotides shows that the dynein regulatory complex is required for cilium motility. Thus cilia-driven flow appears to be a key epigenetic factor in controlling otolith biomineralization, and dynein regulatory complex subunits emerge as candidates for disease genes for ciliopathies in humans.

Date: 2009
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DOI: 10.1038/nature07520

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