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Mutations in Hydin impair ciliary motility in mice

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.

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