1. Developmental Biology
  2. Neuroscience

Exocyst-mediated membrane trafficking of the lissencephaly-associated ECM receptor dystroglycan is required for proper brain compartmentalization

  1. Andriy S Yatsenko
  2. Mariya M Kucherenko
  3. Yuanbin Xie
  4. Henning Urlaub
  5. Halyna R Shcherbata  Is a corresponding author
  1. Hannover Medical School, Germany
  2. Charite Medicine University, Germany
  3. Gannan Medical University, China
  4. University Medical Center Göttingen, Germany
https://doi.org/10.7554/eLife.63868
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Cite this article
  1. Andriy S Yatsenko
  2. Mariya M Kucherenko
  3. Yuanbin Xie
  4. Henning Urlaub
  5. Halyna R Shcherbata
(2021)
Exocyst-mediated membrane trafficking of the lissencephaly-associated ECM receptor dystroglycan is required for proper brain compartmentalization
eLife 10:e63868.
https://doi.org/10.7554/eLife.63868
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Abstract

To assemble a brain, differentiating neurons must make proper connections and establish specialized brain compartments. Abnormal levels of cell adhesion molecules disrupt these processes. Dystroglycan (Dg) is a major non-integrin cell adhesion receptor, deregulation of which is associated with dramatic neuroanatomical defects such as lissencephaly type II, or cobblestone brain. The previously established Drosophila model for cobblestone encephaly was used to understand how Dg is regulated in the brain. During development, Dg has a spatiotemporally dynamic expression pattern, fine-tuning of which is crucial for accurate brain assembly. In addition, mass spectrometry analyses identified numerous components associated with Dg in neurons, including several proteins of the exocyst complex. Data show that exocyst-based membrane trafficking of Dg allows its distinct expression pattern, essential for proper brain morphogenesis. Further studies of the Dg neuronal interactome will allow identification of new factors involved in the development of dystroglycanopathies and advance disease diagnostics in humans.

Data availability

The authors declare that all data supporting the findings of this study are available within the article and its supplementary information files or found on the Dryad Digital Repository (Data DOI: doi:10.5061/dryad.8sf7m0cmf).

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Author details

  1. Andriy S Yatsenko

    Institute of Cell Biochemistry, Hannover Medical School, Hannover, Germany
    Competing interests
    The authors declare that no competing interests exist.

  2. Mariya M Kucherenko

    Institute of Physiology, Charite Medicine University, Berlin, Germany
    Competing interests
    The authors declare that no competing interests exist.

  3. Yuanbin Xie

    Department of Biochemistry and Molecular Biology, School of Basic Medical Science, Gannan Medical University, Ganzhou, China
    Competing interests
    The authors declare that no competing interests exist.

  4. Henning Urlaub

    Bioanalytics, Institute of Clinical Chemistry, University Medical Center Göttingen, Göttingen, Germany
    Competing interests
    The authors declare that no competing interests exist.

  5. Halyna R Shcherbata

    Institute of Cell Biochemistry, Hannover Medical School, Hannover, Germany
    For correspondence
    halyna.shcherbata@mpibpc.mpg.de
    Competing interests
    The authors declare that no competing interests exist.
    ORCID icon "This ORCID iD identifies the author of this article:" 0000-0002-3855-0345

Funding

EMBO (YIP)

  • Halyna R Shcherbata

VW Stiftung (AZN3008)

  • Halyna R Shcherbata

Hannover Medical School

  • Halyna R Shcherbata

VW Stiftung (AZ97750)

  • Halyna R Shcherbata

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

Reviewing Editor

  1. Lucy Erin O'Brien, Stanford University School of Medicine, United States

Version history

  1. Received: October 11, 2020
  2. Accepted: February 23, 2021
  3. Accepted Manuscript published: February 23, 2021 (version 1)
  4. Version of Record published: March 3, 2021 (version 2)

Copyright

© 2021, Yatsenko 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.

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  1. Andriy S Yatsenko
  2. Mariya M Kucherenko
  3. Yuanbin Xie
  4. Henning Urlaub
  5. Halyna R Shcherbata
(2021)
Exocyst-mediated membrane trafficking of the lissencephaly-associated ECM receptor dystroglycan is required for proper brain compartmentalization
eLife 10:e63868.
https://doi.org/10.7554/eLife.63868

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