Behavioral.pdf (2.17 MB)
Behavioral and other phenotypes in a cytoplasmic Dynein light intermediate chain 1 mutant mouse
journal contribution
posted on 2023-06-07, 23:21 authored by Gareth T Banks, Matilda A Haas, Samantha Line, Hazel L Shepherd, Mona AlQatari, Sammy Stewart, Ida Rishal, Amelia Philpott, Majid HafezparastMajid Hafezparast, othersThe 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.
History
Publication status
- Published
File Version
- Published version
Journal
Journal of NeuroscienceISSN
0270-6474Publisher
Society for NeuroscienceExternal DOI
Issue
14Volume
31Page range
5483-5494Department affiliated with
- Neuroscience Publications
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Co-authorFull text available
- Yes
Peer reviewed?
- Yes
Legacy Posted Date
2012-02-06First Open Access (FOA) Date
2016-03-22First Compliant Deposit (FCD) Date
2016-08-17Usage metrics
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