Mutations in Hydin impair ciliary motility in mice.

Mutations in Hydin impair ciliary motility in mice. Research Abstract Details 

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Mutations in Hydin impair ciliary motility in mice. Abstract Text:

Karl-Ferdinand Lechtreck,Philippe Delmotte,Michael L Robinson,Michael J Sanderson,George B Witman,

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.

Mutations in Hydin impair ciliary motility in mice. Publishing Authors By Initials

KF Lechtreck,P Delmotte,ML Robinson,MJ Sanderson,GB Witman,

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Mutations in Hydin impair ciliary motility in mice. Journal Published:

PUBLICATION TYPE: Research Support, Non-U.S. Gov

Journal: The Journal of cell biology

VOLUME: 180

Page Numbers: 633-43

Journal Abbreviation: J. Cell Biol.

ISSN: 1540-8140

DAY: 4

MONTH: 02

YEAR: 2008

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LANGUAGE: eng

NlmUniqueID: 375356

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Grant and Affiliation Information for Mutations in Hydin impair ciliary motility in mice.

AFFILIATION: Department of Cell, University of Massachusetts Medical School, Worcester, MA 01655, USA. Karl.Lechtreck@umassmed.edu

Country: United States

AGENCY: United States NHLBI

GRANT: HL071930

ACRONYM: HL

MEDLINETA: J Cell Biol

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