Centriolar satellites expedite mother centriole remodeling to promote ciliogenesis

Emma A Hall, Dhivya Kumar, Suzanna L Prosser, Patricia L Yeyati, Vicente Herranz-Pérez, Jose Manuel García-Verdugo, Lorraine Rose, Lisa Mckie, Daniel O Dodd, Peter A Tennant, Roly Megaw, Laura C Murphy, Marisa F Ferreira, Graeme Grimes, Lucy Williams, Tooba Quidwai, Laurence Pelletier, Jeremy F Reiter, Pleasantine Mill

Research output: Contribution to journalArticlepeer-review


Centrosomes are orbited by centriolar satellites, dynamic multiprotein assemblies nucleated by Pericentriolar Material 1 (PCM1). To study the requirement for centriolar satellites, we generated mice lacking PCM1, a crucial component of satellites. Pcm1-/- mice display partially penetrant perinatal lethality with survivors exhibiting hydrocephalus, oligospermia and cerebellar hypoplasia, and variably expressive phenotypes such as hydronephrosis. As many of these phenotypes have been observed in human ciliopathies and satellites are implicated in cilia biology, we investigated whether cilia were affected. PCM1 was dispensable for ciliogenesis in many cell types, whereas Pcm1-/- multiciliated ependymal cells and human PCM1-/- retinal pigmented epithelial 1 (RPE1) cells showed reduced ciliogenesis. PCM1-/- RPE1 cells displayed reduced docking of the mother centriole to the ciliary vesicle and removal of CP110 and CEP97 from the distal mother centriole, indicating compromised early ciliogenesis. Similarly, Pcm1-/- ependymal cells exhibited reduced removal of CP110 from basal bodies in vivo. We propose that PCM1 and centriolar satellites facilitate efficient trafficking of proteins to and from centrioles, including the departure of CP110 and CEP97 to initiate ciliogenesis, and that the threshold to trigger ciliogenesis differs between cell types.
Original languageEnglish
Article numbere79299
Early online date15 Feb 2023
Publication statusPublished - 15 Feb 2023


  • Animals
  • Cell Cycle Proteins/genetics
  • Centrioles/metabolism
  • Centrosome/metabolism
  • Cilia/metabolism
  • Cytoskeletal Proteins/metabolism
  • Female
  • Humans
  • Mice
  • Mothers


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