ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2011
Behavioral and other phenotypes in a cytoplasmic Dynein light intermediate chain 1 mutant mouse.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.
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Who cites it
13 citing papers in PubMed, 26 citations in OpenAlex.
- The roles of cytoplasmic dynein complex in various ocular disorders.Molecular medicine (Cambridge, Mass.) · 2026Review
- Non-transport functions of motor proteins in corticogenesis.Frontiers in cell and developmental biology · 2026Review
- Large scale similarity search across digital reconstructions of neural morphology.Neuroscience research · 2022Article
- Dync1li1 is required for the survival of mammalian cochlear hair cells by regulating the transportation of autophagosomes.PLoS genetics · 2022Article
- Association of DYNC1H1 gene SNP/CNV with disease susceptibility, GCs efficacy, HRQOL, anxiety, and depression in Chinese SLE patients.Journal of clinical laboratory analysis · 2021Article
- Proteomics analysis of proteins interacting with heat shock factor 1 in squamous cell carcinoma of the cervix.Oncology letters · 2019Article
- Sharing Neuron Data: Carrots, Sticks, and Digital Records.PLoS biology · 2015Article
- Cytoplasmic dynein and its regulatory proteins in Golgi pathology in nervous system disorders.Frontiers in neuroscience · 2015Review
- Analysis of dynein intermediate chains, light intermediate chains and light chains in a cohort of hereditary peripheral neuropathies.Neurogenetics · 2014Article
- Dlic1 deficiency impairs ciliogenesis of photoreceptors by destabilizing dynein.Cell research · 2013Article
- Establishing a novel knock-in mouse line for studying neuronal cytoplasmic dynein under normal and pathologic conditions.Cytoskeleton (Hoboken, N.J.) · 2013Article
- Article
- Visualization and genetic manipulation of dendrites and spines in the mouse cerebral cortex and hippocampus using in utero electroporation.Journal of visualized experiments : JoVE · 2012Article
Corrections and comments
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Authors and funding
21 authors at 7 institutions in 3 countries.
Funding
Abstract
The cytoplasmic dynein complex is fundamentally important to all eukaryotic cells for transporting a variety of essential cargoes along microtubules within the cell. This complex also plays more specialized roles in neurons. The complex consists of 11 types of protein that interact with each other and with external adaptors, regulators and cargoes. Despite the importance of the cytoplasmic dynein complex, we know comparatively little of the roles of each component protein, and in mammals few mutants exist that allow us to explore the effects of defects in dynein-controlled processes in the context of the whole organism. Here we have taken a genotype-driven approach in mouse (Mus musculus) to analyze the role of one subunit, the dynein light intermediate chain 1 (Dync1li1). We find that, surprisingly, an N235Y point mutation in this protein results in altered neuronal development, as shown from in vivo studies in the developing cortex, and analyses of electrophysiological function. Moreover, mutant mice display increased anxiety, thus linking dynein functions to a behavioral phenotype in mammals for the first time. These results demonstrate the important role that dynein-controlled processes play in the correct development and function of the mammalian nervous system.
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Registered trials
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.