FAP57/WDR65 targets assembly of a subset of inner arm dyneins and connects to regulatory hubs in cilia.

ABSTRACT: Ciliary motility depends on both the precise spatial organization of multiple dynein motors within the 96 nm axonemal repeat and the highly coordinated interactions between different dyneins and regulatory complexes located at the base of the radial spokes. Mutations in genes encoding cytoplasmic assembly factors, intraflagellar transport factors, docking proteins, dynein subunits, and associated regulatory proteins can all lead to defects in dynein assembly and ciliary motility. Significant progress has been made in the identification of dynein subunits and extrinsic factors required for preassembly of dynein complexes in the cytoplasm, but less is known about the docking factors that specify the unique binding sites for the different dynein isoforms on the surface of the doublet microtub

SUBMITTER: Lin J 

PROVIDER: S-EPMC6761771 | biostudies-literature | 2019 Oct

REPOSITORIES: biostudies-literature