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

Yatsenko, A. S., Kucherenko, M. M., Xie, Y., Urlaub, H., & Shcherbata, H. R. (2021). Exocyst-mediated membrane trafficking of the lissencephaly-associated ECM receptor dystroglycan is required for proper brain compartmentalization. eLife, 10: e63868. doi:10.7554/eLife.63868.

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Yatsenko, A. S.1, Author              
Kucherenko, M. M.1, Author              
Xie, Y., Author
Urlaub, H.2, Author              
Shcherbata, H. R.1, Author              
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1Research Group of Gene Expression and Signaling, MPI for biophysical chemistry, Max Planck Society, ou_578608              
2Research Group of Bioanalytical Mass Spectrometry, MPI for biophysical chemistry, Max Planck Society, ou_578613              

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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 lissencephaly 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.

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Language(s): eng - English
 Dates: 2021-02-23
 Publication Status: Published online
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 Rev. Type: Peer
 Identifiers: DOI: 10.7554/eLife.63868
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Title: eLife
Source Genre: Journal
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Pages: - Volume / Issue: 10 Sequence Number: e63868 Start / End Page: - Identifier: -