ArticleNeural development2012
Schwann cell myelination requires Dynein function.
Article in Neural development, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed.
- Molecular Motors in Myelination and Their Misregulation in Disease.Molecular neurobiology · 2025Review
- The expanding clinical and genetic spectrum of DYNC1H1-related disorders.Brain : a journal of neurology · 2025Article
- Patient-specific mutation of Dync1h1 in mice causes brain and behavioral deficits.Neurobiology of disease · 2024Article
- Article
- Spinal cord precursors utilize neural crest cell mechanisms to generate hybrid peripheral myelinating glia.eLife · 2021Article
- Zebrafish is a central model to dissect the peripheral neuropathy.Genes & genomics · 2019Review
- Dynein promotes sustained axonal growth and Schwann cell remodeling early during peripheral nerve regeneration.PLoS genetics · 2019Article
- Axonal transport in a peripheral diabetic neuropathy model: sex-dimorphic features.Biology of sex differences · 2018Article
- Dynein/dynactin is necessary for anterograde transport ofProceedings of the National Academy of Sciences of the United States of America · 2017Article
- Analysis of myelinated axon formation in zebrafish.Methods in cell biology · 2017Review
- Pharmacological treatment and BBB-targeted genetic therapy for MCT8-dependent hypomyelination in zebrafish.Disease models & mechanisms · 2016Article
- Fbxw7 Limits Myelination by Inhibiting mTOR Signaling.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2015Article
- Mutation screen reveals novel variants and expands the phenotypes associated with DYNC1H1.Journal of neurology · 2015Article
- Review
- CNS myelination requires cytoplasmic dynein function.Developmental dynamics : an official publication of the American Association of Anatomists · 2015Article
- Motoneuron development influences dorsal root ganglia survival and Schwann cell development in a vertebrate model of spinal muscular atrophy.Human molecular genetics · 2015Article
- Transplantation of neurotrophin-3-transfected bone marrow mesenchymal stem cells for the repair of spinal cord injury.Neural regeneration research · 2014Article
- In vivo cell biology in zebrafish - providing insights into vertebrate development and disease.Journal of cell science · 2014Review
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Authors and funding
2 authors.
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Abstract
backgroundInteraction of Schwann cells with axons triggers signal transduction that drives expression of Pou3f1 and Egr2 transcription factors, which in turn promote myelination. Signal transduction appears to be mediated, at least in part, by cyclic adenosine monophosphate (cAMP) because elevation of cAMP levels can stimulate myelination in the absence of axon contact. The mechanisms by which the myelinating signal is conveyed remain unclear.
resultsBy analyzing mutations that disrupt myelination in zebrafish, we learned that Dynein cytoplasmic 1 heavy chain 1 (Dync1h1), which functions as a motor for intracellular molecular trafficking, is required for peripheral myelination. In dync1h1 mutants, Schwann cell progenitors migrated to peripheral nerves but then failed to express Pou3f1 and Egr2 or make myelin membrane. Genetic mosaic experiments revealed that robust Myelin Basic Protein expression required Dync1h1 function within both Schwann cells and axons. Finally, treatment of dync1h1 mutants with a drug to elevate cAMP levels stimulated myelin gene expression.
conclusionDync1h1 is required for retrograde transport in axons and mutations of Dync1h1 have been implicated in axon disease. Our data now provide evidence that Dync1h1 is also required for efficient myelination of peripheral axons by Schwann cells, perhaps by facilitating signal transduction necessary for myelination.
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