Abstract
Genetic analysis in model organisms and human patient populations has revealed that a surprisingly large number of genes are needed to get axonemal dyneins into cilia, where they power the motility of these essential organelles. Most of these genes encode unique axonemal dynein assembly factors that are conserved among most eukaryotes that have motile cilia, whereas comparable assembly factors do not appear necessary for cytoplasmic dynein function. The known roles of assembly factors include cytoplasmic preassembly by chaperones, intraflagellar transport-dependent transport into cilia, and docking to specific sites on ciliary doublet microtubules. Understanding how these gene products work is important both to unravel general ciliary protein assembly mechanisms and to explain the etiology of many cases of human primary ciliary dyskinesia, the majority of which result from dynein assembly defects.
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