The hydrocephalus inducing gene product, Hydin, positions axonemal central pair microtubules.

ABSTRACT:

Background

Impairment of cilia and flagella function underlies a growing number of human genetic diseases. Mutations in hydin in hy3 mice cause lethal communicating hydrocephalus with early onset. Hydin was recently identified as an axonemal protein; however, its function is as yet unknown.

Results

Here we use RNAi in Trypanosoma brucei to address this issue and demonstrate that loss of Hydin causes slow growth and a loss of cell motility. We show that two separate defects in newly-formed flagellar central pair microtubules underlie the loss of cell motility. At early time-points after RNAi induction, the central pair becomes mispositioned, while at later time points the central pair is lost. While the basal body is unaffected, both defects originate at the basal plate, refl

SUBMITTER: Dawe HR 

PROVIDER: S-EPMC2048497 | biostudies-literature | 2007 Aug

REPOSITORIES: biostudies-literature