ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2015
CNS myelination requires cytoplasmic dynein function.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
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Who cites it
16 citing papers in PubMed, 28 citations in OpenAlex.
- Label-free and fluorescence imaging of oligodendrocytes and myelin.Npj imaging · 2025Review
- Molecular Motors in Myelination and Their Misregulation in Disease.Molecular neurobiology · 2025Review
- Patient-specific mutation of Dync1h1 in mice causes brain and behavioral deficits.Neurobiology of disease · 2024Article
- Pro-myelinating clemastine administration improves recording performance of chronically implanted microelectrodes and nearby neuronal health.Biomaterials · 2023Article
- Requirement of Zebrafish Adcy3a and Adcy5 in Melanosome Dispersion and Melanocyte Stripe Formation.International journal of molecular sciences · 2022Article
- Whole-exome sequencing identifies a novel de novo variant in DYNC1H in a patient with intractable epilepsy.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2022Review
- Missense mutation inWorld journal of clinical cases · 2021Article
- Insights Into Central Nervous System Glial Cell Formation and Function From Zebrafish.Frontiers in cell and developmental biology · 2021Review
- Comparative Proteomic Profiling Identifies Reciprocal Expression of Mitochondrial Proteins Between White and Gray Matter Lesions From Multiple Sclerosis Brains.Frontiers in neurology · 2021Article
- The clinical-phenotype continuum in DYNC1H1-related disorders-genomic profiling and proposal for a novel classification.Journal of human genetics · 2020Article
- Mechanistic Target of Rapamycin Regulates the Oligodendrocyte Cytoskeleton during Myelination.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2020Article
- Article
- NDE1 positively regulates oligodendrocyte morphological differentiation.Scientific reports · 2018Article
- Dynein/dynactin is necessary for anterograde transport ofProceedings of the National Academy of Sciences of the United States of America · 2017Article
- Genomic and phenotypic signatures of climate adaptation in anEcology and evolution · 2017Article
- Analysis of myelinated axon formation in zebrafish.Methods in cell biology · 2017Review
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Authors and funding
7 authors at 1 institution in 1 country.
Funding
Abstract
backgroundCytoplasmic dynein provides the main motor force for minus-end-directed transport of cargo on microtubules. Within the vertebrate central nervous system (CNS), proliferation, neuronal migration, and retrograde axon transport are among the cellular functions known to require dynein. Accordingly, mutations of DYNC1H1, which encodes the heavy chain subunit of cytoplasmic dynein, have been linked to developmental brain malformations and axonal pathologies. Oligodendrocytes, the myelinating glial cell type of the CNS, migrate from their origins to their target axons and subsequently extend multiple long processes that ensheath axons with specialized insulating membrane. These processes are filled with microtubules, which facilitate molecular transport of myelin components. However, whether oligodendrocytes require cytoplasmic dynein to ensheath axons with myelin is not known.
resultsWe identified a mutation of zebrafish dync1h1 in a forward genetic screen that caused a deficit of oligodendrocytes. Using in vivo imaging and gene expression analyses, we additionally found evidence that dync1h1 promotes axon ensheathment and myelin gene expression.
conclusionsIn addition to its well known roles in axon transport and neuronal migration, cytoplasmic dynein contributes to neural development by promoting myelination.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.