Alstrom syndrome gene is a stem cell-specific regulator of centriole duplication in the Drosophila testis

  1. Cuie Chen
  2. Yukiko M Yamashita  Is a corresponding author
  1. University of Michigan, United States
  2. Whitehead Institute/MIT, United States


Asymmetrically dividing stem cells often show asymmetric behavior of the mother versus daughter centrosomes, whereby the self-renewing stem cell selectively inherits the mother or daughter centrosome. Although the asymmetric centrosome behavior is widely conserved, its biological significance remains largely unclear. Here we show that Alms1a, a Drosophila homolog of the human ciliopathy gene Alstrom syndrome, is enriched on the mother centrosome in Drosophila male germline stem cells (GSCs). Depletion of alms1a in GSCs, but not in differentiating germ cells, results in rapid loss of centrosomes due to a failure in daughter centriole duplication, suggesting that Alms1a has a stem cell-specific function in centrosome duplication. Alms1a interacts with Sak/Plk4, a critical regulator of centriole duplication, more strongly at the GSC mother centrosome, further supporting Alms1a's unique role in GSCs. Our results begin to reveal the unique regulation of stem cell centrosomes that may contribute to asymmetric stem cell divisions.

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All data generated or analysed during this study are included in the manuscript and supporting files.

Article and author information

Author details

  1. Cuie Chen

    Life Sciences Institute, University of Michigan, Ann Arbor, United States
    Competing interests
    No competing interests declared.
    ORCID icon "This ORCID iD identifies the author of this article:" 0000-0002-5498-9753
  2. Yukiko M Yamashita

    Biology, Whitehead Institute/MIT, Cambridge, United States
    For correspondence
    Competing interests
    Yukiko M Yamashita, Reviewing editor, eLife.
    ORCID icon "This ORCID iD identifies the author of this article:" 0000-0001-5541-0216


Howard Hughes Medical Institute

  • Yukiko M Yamashita

National Institute of General Medical Sciences (R01GM118308)

  • Yukiko M Yamashita

The funders had no role in study design, data collection and interpretation, or the decision to submit the work for publication.

Reviewing Editor

  1. Anna Akhmanova, Utrecht University, Netherlands

Version history

  1. Received: May 27, 2020
  2. Accepted: September 22, 2020
  3. Accepted Manuscript published: September 23, 2020 (version 1)
  4. Version of Record published: October 5, 2020 (version 2)


© 2020, Chen & Yamashita

This article is distributed under the terms of the Creative Commons Attribution License permitting unrestricted use and redistribution provided that the original author and source are credited.


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  1. Cuie Chen
  2. Yukiko M Yamashita
Alstrom syndrome gene is a stem cell-specific regulator of centriole duplication in the Drosophila testis
eLife 9:e59368.

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