1. Cell Biology
  2. Developmental Biology and Stem Cells
Download icon

Flattop regulates basal body docking and positioning in mono- and multiciliated cells

  1. Moritz Gegg
  2. Anika Böttcher
  3. Ingo Burtscher
  4. Stefan Hasenöder
  5. Claude Van Campenhout
  6. Michaela Aichler
  7. Axel Walch
  8. Seth G. N. Grant
  9. Heiko Lickert Is a corresponding author
  1. Helmholtz Center Munich, Germany
  2. Université libre de Bruxelles, Belgium
  3. University of Edinburgh, United Kingdom
Research Article
Cited
10
Views
2,502
Comments
0
Cite as: eLife 2014;3:e03842 doi: 10.7554/eLife.03842

Abstract

Planar cell polarity (PCP) regulates basal body (BB) docking and positioning during cilia formation, but the underlying mechanisms remain elusive. Here, we investigate the uncharacterized gene Flattop (Fltp) that is transcriptionally activated during PCP acquisition in ciliated tissues. Fltp knock-out mice show BB docking and ciliogenesis defects in multiciliated lung cells. Furthermore, Fltp is necessary for kinocilium positioning in monociliated inner ear hair cells. In these cells, the core PCP molecule Dishevelled 2, the BB/spindle positioning protein Dlg3 and Fltp localize directly adjacent at the apical plasma membrane, physically interact and surround the BB at the interface of the microtubule and actin cytoskeleton. Dlg3 and Fltp knock-outs suggest that both cooperatively translate PCP cues for BB positioning in the inner ear. Taken together, the identification of novel BB/spindle positioning components as potential mediators of PCP signaling might have broader implications for other cell types, ciliary disease and asymmetric cell division.

Article and author information

Author details

  1. Moritz Gegg

    1. Helmholtz Center Munich, Munich, Germany
    Competing interests
    The authors declare that no competing interests exist.
  2. Anika Böttcher

    1. Helmholtz Center Munich, Munich, Germany
    Competing interests
    The authors declare that no competing interests exist.
  3. Ingo Burtscher

    1. Helmholtz Center Munich, Munich, Germany
    Competing interests
    The authors declare that no competing interests exist.
  4. Stefan Hasenöder

    1. Helmholtz Center Munich, Munich, Germany
    Competing interests
    The authors declare that no competing interests exist.
  5. Claude Van Campenhout

    1. Université libre de Bruxelles, Gosselies, Belgium
    Competing interests
    The authors declare that no competing interests exist.
  6. Michaela Aichler

    1. Helmholtz Center Munich, Munich, Germany
    Competing interests
    The authors declare that no competing interests exist.
  7. Axel Walch

    1. Helmholtz Center Munich, Munich, Germany
    Competing interests
    The authors declare that no competing interests exist.
  8. Seth G. N. Grant

    1. University of Edinburgh, Edinburgh, United Kingdom
    Competing interests
    The authors declare that no competing interests exist.
  9. Heiko Lickert

    1. Helmholtz Center Munich, Munich, Germany
    For correspondence
    1. heiko.lickert@helmholtz-muenchen.de
    Competing interests
    The authors declare that no competing interests exist.

Ethics

Animal experimentation: Mouse keeping was done at the central facilities at HMGU in accordance with the German animal welfare legislation and acknowledged guidelines of the Society of Laboratory Animals (GV-SOLAS) and of the Federation of Laboratory Animal Science Associations (FELASA). Post-mortem examination of organs was not subject to regulatory authorization.

Reviewing Editor

  1. Jeremy Nathans, Reviewing Editor, Howard Hughes Medical Institute, Johns Hopkins University School of Medicine, United States

Publication history

  1. Received: July 1, 2014
  2. Accepted: October 7, 2014
  3. Accepted Manuscript published: October 8, 2014 (version 1)
  4. Version of Record published: November 6, 2014 (version 2)

Copyright

© 2014, Gegg et al.

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.

Metrics

  • 2,502
    Page views
  • 348
    Downloads
  • 10
    Citations

Article citation count generated by polling the highest count across the following sources: Crossref, Scopus, PubMed Central.

Comments

Download links

A two-part list of links to download the article, or parts of the article, in various formats.

Downloads (link to download the article as PDF)

Download citations (links to download the citations from this article in formats compatible with various reference manager tools)

Open citations (links to open the citations from this article in various online reference manager services)

Further reading

    1. Developmental Biology and Stem Cells
    Cyrille Ramond et al.
    Research Article