Towards an atomic model of a beating ciliary axoneme

The axoneme of motile cilia and eukaryotic flagella is an ordered assembly of hundreds of proteins that powers the locomotion of single cells and generates flow of liquid and particles across certain mammalian tissues. The symmetric and organized structure of the axoneme has invited structural biologists to unravel its intricate architecture at different scales. In the last few years, single-particle cryo-electron microscopy provided high-resolution structures of axonemal complexes that comprise dozens of proteins and are key to cilia function. This review summarizes unique structural features of the axoneme and the framework they provide to understand cilia assembly, the mechanism of ciliary beating, and clinical conditions associated with impaired cilia motility.