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Published online
doi:10.1083/jcb.200710162
The Journal of Cell Biology, Vol. 180, No. 3, 633-643
The Rockefeller University Press, 0021-9525 $30.00
© Lechtreck et al.
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Article

Mutations in Hydin impair ciliary motility in mice



Karl-Ferdinand Lechtreck1, Philippe Delmotte2, Michael L. Robinson3, Michael J. Sanderson2, and George B. Witman1

1 Department of Cell Biology and 2 Department of Physiology, University of Massachusetts Medical School, Worcester, MA 01655
3 Zoology Department, Miami University, Oxford, OH 45056

Correspondence to K.-F. Lechtreck: Karl.Lechtreck{at}umassmed.edu; or G.B. Witman: George.Witman{at}umassmed.edu

Chlamydomonas reinhardtii hydin is a central pair protein required for flagellar motility, and mice with Hydin defects develop lethal hydrocephalus. To determine if defects in Hydin cause hydrocephalus through a mechanism involving cilia, we compared the morphology, ultrastructure, and activity of cilia in wild-type and hydin mutant mice strains. The length and density of cilia in the brains of mutant animals is normal. The ciliary axoneme is normal with respect to the 9 + 2 microtubules, dynein arms, and radial spokes but one of the two central microtubules lacks a specific projection. The hydin mutant cilia are unable to bend normally, ciliary beat frequency is reduced, and the cilia tend to stall. As a result, these cilia are incapable of generating fluid flow. Similar defects are observed for cilia in trachea. We conclude that hydrocephalus in hydin mutants is caused by a central pair defect impairing ciliary motility and fluid transport in the brain.

Abbreviations used in this paper: CBF, ciliary beat frequency; CP, central pair; P, postnatal day; PCD, primary ciliary dyskinesia; SEM, scanning EM; Spag6, sperm-associated antigen 6; TEM, transmission EM.


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