| |  Online-Ressource |
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| Verfasst von: | Medina-Cano, Daniel [VerfasserIn]  |
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| | Ucuncu, Ekin [VerfasserIn] |
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| | Nguyen, Lam Son [VerfasserIn] |
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| | Nicouleau, Michael [VerfasserIn] |
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| | Lipecka, Joanna [VerfasserIn] |
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| | Bizot, Jean-Charles [VerfasserIn] |
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| | Thiel, Christian [VerfasserIn] |
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| | Foulquier, François [VerfasserIn] |
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| | Lefort, Nathalie [VerfasserIn] |
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| | Faivre-Sarrailh, Catherine [VerfasserIn] |
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| | Colleaux, Laurence [VerfasserIn] |
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| | Guerrera, Ida Chiara [VerfasserIn] |
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| | Cantagrel, Vincent [VerfasserIn] |
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| Titel: | High N-glycan multiplicity is critical for neuronal adhesion and sensitizes the developing cerebellum to N-glycosylation defect |
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| Verf.angabe: | Daniel Medina-Cano, Ekin Ucuncu, Lam Son Nguyen, Michael Nicouleau, Joanna Lipecka, Jean-Charles Bizot, Christian Thiel, François Foulquier, Nathalie Lefort, Catherine Faivre-Sarrailh, Laurence Colleaux, Ida Chiara Guerrera, Vincent Cantagrel |
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| E-Jahr: | 2018 |
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| Jahr: | 12 October 2018 |
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| Umfang: | 27 S. |
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| Fussnoten: | Gesehen am 25.03.2020 |
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| Titel Quelle: | Enthalten in: eLife |
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| Ort Quelle: | Cambridge : eLife Sciences Publications, 2012 |
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| Jahr Quelle: | 2018 |
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| Band/Heft Quelle: | 7(2018) Artikel-Nummer e38309, 27 Seiten |
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| ISSN Quelle: | 2050-084X |
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| Abstract: | Proper brain development relies highly on protein N-glycosylation to sustain neuronal migration, axon guidance and synaptic physiology. Impairing the N-glycosylation pathway at early steps produces broad neurological symptoms identified in congenital disorders of glycosylation. However, little is known about the molecular mechanisms underlying these defects. We generated a cerebellum specific knockout mouse for Srd5a3, a gene involved in the initiation of N-glycosylation. In addition to motor coordination defects and abnormal granule cell development, Srd5a3 deletion causes mild N-glycosylation impairment without significantly altering ER homeostasis. Using proteomic approaches, we identified that Srd5a3 loss affects a subset of glycoproteins with high N-glycans multiplicity per protein and decreased protein abundance or N-glycosylation level. As IgSF-CAM adhesion proteins are critical for neuron adhesion and highly N-glycosylated, we observed impaired IgSF-CAM-mediated neurite outgrowth and axon guidance in Srd5a3 mutant cerebellum. Our results link high N-glycan multiplicity to fine-tuned neural cell adhesion during mammalian brain development. |
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| DOI: | doi:10.7554/eLife.38309 |
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| URL: | Bitte beachten Sie: Dies ist ein Bibliographieeintrag. Ein Volltextzugriff für Mitglieder der Universität besteht hier nur, falls für die entsprechende Zeitschrift/den entsprechenden Sammelband ein Abonnement besteht oder es sich um einen OpenAccess-Titel handelt.
Volltext: https://doi.org/10.7554/eLife.38309 |
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| | DOI: https://doi.org/10.7554/eLife.38309 |
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| Datenträger: | Online-Ressource |
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| Sprache: | eng |
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| Sach-SW: | 3-Oxo-5-alpha-Steroid 4-Dehydrogenase |
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| | Animals |
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| | Axon Guidance |
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| | cell adhesion |
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| | Cell Adhesion |
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| | Cell Adhesion Molecules |
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| | cell biology |
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| | Cell Differentiation |
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| | Cell Membrane |
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| | cerebellum |
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| | Cerebellum |
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| | congenital disorders of glycosylation |
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| | Cytoplasmic Granules |
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| | Gene Deletion |
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| | Glycosylation |
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| | human |
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| | Immunoglobulins |
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| | Induced Pluripotent Stem Cells |
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| | Membrane Proteins |
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| | Mice, Knockout |
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| | Motor Activity |
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| | mouse |
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| | Mutation |
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| | N-glycosylation |
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| | Neural Pathways |
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| | neuronal migration |
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| | Neurons |
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| | neuroscience |
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| | Polysaccharides |
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| | proteomics |
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| | Proteomics |
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| | Purkinje Cells |
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| | Reproducibility of Results |
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| | Unfolded Protein Response |
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| K10plus-PPN: | 1693262355 |
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| Verknüpfungen: | → Zeitschrift |
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High N-glycan multiplicity is critical for neuronal adhesion and sensitizes the developing cerebellum to N-glycosylation defect / Medina-Cano, Daniel [VerfasserIn]; 12 October 2018 (Online-Ressource)
