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SFI1 promotes centriole duplication by recruiting USP9X to stabilize the microcephaly protein STIL

Kodani, Andrew, Moyer, Tyler, Chen, Allen, Holland, Andrew, Walsh, Christopher A., Reiter, Jeremy F.·2019·Journal of Cell Biology
DOI 10.1083/jcb.201803041
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Abstract

In mammals, centrioles participate in brain development, and human mutations affecting centriole duplication cause microcephaly. Here, we identify a role for the mammalian homologue of yeast SFI1, involved in the duplication of the yeast spindle pole body, as a critical regulator of centriole duplication in mammalian cells. Mammalian SFI1 interacts with USP9X, a deubiquitylase associated with human syndromic mental retardation. SFI1 localizes USP9X to the centrosome during S phase to deubiquitylate STIL, a critical regulator of centriole duplication. USP9X-mediated deubiquitylation protects STIL from degradation. Consistent with a role for USP9X in stabilizing STIL, cells from patients with USP9X loss-of-function mutations have reduced STIL levels. Together, these results demonstrate that SFI1 is a centrosomal protein that localizes USP9X to the centrosome to stabilize STIL and promote centriole duplication. We propose that the USP9X protection of STIL to facilitate centriole duplication underlies roles of both proteins in human neurodevelopment.

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  • Disruption of endosomal trafficking phenocopies CQ-induced fusion blockade and exacerbates neurodegeneration.

    “disruption of endosomal trafficking phenocopies CQ-induced fusion blockade and exacerbates neurodegeneration (Kodani et al. 2019).”

    From TMEM55B downregulation promotes neurodegeneration in Alzheimer’s disease models (2026) · cites this paper as Kodani et al. 2019

  • Chloroquine primarily blocks autophagic flux by impairing autophagosome-lysosome fusion through disruption of endo-lysosomal trafficking pathways and vesicular fusion machinery.

    “Two landmark investigations, Mauthe et al., 2018 andNnah et al., 2019, demonstrated that CQ primarily blocks autophagic flux by impairing autophagosome-lysosome fusion through disruption of endo-lysosomal trafficking pathways and vesicular fusion machinery (Mauthe et al. 2018), (Kodani et al. 2019).”

    From TMEM55B downregulation promotes neurodegeneration in Alzheimer’s disease models (2026) · cites this paper as Kodani et al. 2019

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Metadata

Authors
Kodani, A., Moyer, T., Chen, A., Holland, A., Walsh, C. A., & Reiter, J. F.
Affiliations
  • Department of Biochemistry and Biophysics, University of California, San Francisco, San Francisco, CA 1
  • Department of Molecular Biology and Genetics, Johns Hopkins University School of Medicine, Baltimore, MD 3
  • Division of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children’s Hospital, Boston, MA 4
ORCID
Venue
Journal of Cell Biology
Publisher
Rockefeller University Press
Published
2019-06-13
Volume
218
Issue
7
Pages
2185-2197
ISSN
  • 0021-9525
  • 1540-8140
DOI
10.1083/jcb.201803041
Cited by
25
References
2
Language
en
Licence
Funders
  • Charles Hood Foundation
  • William Randolph Hearst Fund
  • National Institutes of Health (1R21NS104633-01A1, R01 GM114119, R01 AR054396, GM095941)
  • American Cancer Society (129742-RSG-16-156001-CCG)
  • National Institute of Neurological Disorders and Stroke (R01 NS035129, R01 NS32457)
  • Manton Center for Orphan Disease Research
  • Howard Hughes Medical Institute

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