RT Journal Article SR Electronic T1 Sub-centrosomal mapping identifies augmin-γTuRC as part of a centriole-stabilizing scaffold JF bioRxiv FD Cold Spring Harbor Laboratory SP 2020.11.18.384156 DO 10.1101/2020.11.18.384156 A1 Nina Schweizer A1 Laurence Haren A1 Ricardo Viais A1 Cristina Lacasa A1 Ilaria Dutto A1 Andreas Merdes A1 Jens Lüders YR 2020 UL http://biorxiv.org/content/early/2020/11/18/2020.11.18.384156.abstract AB Centriole biogenesis and maintenance are crucial for cells to generate cilia and assemble centrosomes that function as microtubule organizing centers (MTOCs). Centriole biogenesis and MTOC function both require the microtubule nucleator γ-tubulin ring complex (γTuRC). The widely accepted view is that γTuRC localizes to the pericentriolar material (PCM), where it nucleates microtubules. γTuRC has also been observed at centriolar regions that lack PCM, but the significance of these findings is unclear. Here we have used expansion microscopy to map spatially and functionally distinct sub-populations of centrosomal γTuRC including in the centriole lumen. Luminal localization is mediated by augmin and both complexes are linked to the centriole inner scaffold through POC5. Disruption of luminal localization impairs centriole stability and cilia assembly, defects that are also observed in γTuRC mutant fibroblasts derived from a patient suffering from microcephaly with chorioretinopathy. These results identify a novel, non-canonical role of augmin-γTuRC in the centriole lumen that is linked to human disease.Competing Interest StatementThe authors have declared no competing interest.