Centriolar satellites expedite mother centriole remodeling to promote ciliogenesis

• Published in: eLife Vol. 12
• Main Authors: Hall, Emma A, Kumar, Dhivya, Prosser, Suzanna L, Yeyati, Patricia L, Herranz-Pérez, Vicente, García-Verdugo, Jose Manuel, Rose, Lorraine, McKie, Lisa, Dodd, Daniel O, Tennant, Peter A, Megaw, Roly, Murphy, Laura C, Ferreira, Marisa F, Grimes, Graeme, Williams, Lucy, Quidwai, Tooba, Pelletier, Laurence, Reiter, Jeremy F, Mill, Pleasantine
• Format: Journal Article
• Language: English
• Published: England eLife Science Publications, Ltd 15-02-2023; eLife Sciences Publications, Ltd; eLife Sciences Publications Ltd
• Subjects: Animals; Artificial satellites; Cell Cycle Proteins - genetics; Cell Cycle Proteins - metabolism; centriolar satellites; centriole; Centrioles - metabolism; Centrosome - metabolism; cilia; Cilia - metabolism; ciliary vesicle; ciliogenesis; condensates; Cytoskeletal Proteins - metabolism; Developmental Biology; Female; Humans; Mice; Photographic industry; Proteins; Scientific equipment and supplies industry; United States; Japan; mouse; ciliary vesicle; ciliogenesis; centriolar satellites; developmental biology; cilia; condensates; centriole; centrosome; human
• ISSN: 2050-084X; 2050-084X
• DOI: 10.7554/elife.79299

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.
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
multiciliated ependymal cells and human
retinal pigmented epithelial 1 (RPE1) cells showed reduced ciliogenesis.
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,
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.