Microtubule asters anchored by FSD1 control axoneme assembly and ciliogenesis
Open Access
- 11 December 2018
- journal article
- research article
- Published by Springer Science and Business Media LLC in Nature Communications
- Vol. 9 (1), 5277
- https://doi.org/10.1038/s41467-018-07664-2
Abstract
Defective ciliogenesis causes human developmental diseases termed ciliopathies. Microtubule (MT) asters originating from centrosomes in mitosis ensure the fidelity of cell division by positioning the spindle apparatus. However, the function of microtubule asters in interphase remains largely unknown. Here, we reveal an essential role of MT asters in transition zone (TZ) assembly during ciliogenesis. We demonstrate that the centrosome protein FSD1, whose biological function is largely unknown, anchors MT asters to interphase centrosomes by binding to microtubules. FSD1 knockdown causes defective ciliogenesis and affects embryonic development in vertebrates. We further show that disruption of MT aster anchorage by depleting FSD1 or other known anchoring proteins delocalizes the TZ assembly factor Cep290 from centriolar satellites, and causes TZ assembly defects. Thus, our study establishes FSD1 as a MT aster anchorage protein and reveals an important function of MT asters anchored by FSD1 in TZ assembly during ciliogenesis.This publication has 66 references indexed in Scilit:
- The ciliary transition zone: from morphology and molecules to medicineTrends in Cell Biology, 2012
- Small-molecule inhibitors of the AAA+ ATPase motor cytoplasmic dyneinNature, 2012
- A transition zone complex regulates mammalian ciliogenesis and ciliary membrane compositionNature Genetics, 2011
- Mapping the NPHP-JBTS-MKS Protein Network Reveals Ciliopathy Disease Genes and PathwaysCell, 2011
- Nde1-mediated inhibition of ciliogenesis affects cell cycle re-entryNature, 2011
- CEP290 tethers flagellar transition zone microtubules to the membrane and regulates flagellar protein contentThe Journal of cell biology, 2010
- The Vertebrate Primary Cilium in Development, Homeostasis, and DiseaseCell, 2009
- The Human Kinetochore Ska1 Complex Facilitates Microtubule Depolymerization-Coupled MotilityDevelopmental Cell, 2009
- Odf2-deficient mother centrioles lack distal/subdistal appendages and the ability to generate primary ciliaNature, 2005
- Dynamics and mechanics of the microtubule plus endNature, 2003