RT Journal Article
SR Electronic
T1 Behavioral and Other Phenotypes in a Cytoplasmic Dynein Light Intermediate Chain 1 Mutant Mouse
JF The Journal of Neuroscience
JO J. Neurosci.
FD Society for Neuroscience
SP 5483
OP 5494
DO 10.1523/JNEUROSCI.5244-10.2011
VO 31
IS 14
A1 Gareth T. Banks
A1 Matilda A. Haas
A1 Samantha Line
A1 Hazel L. Shepherd
A1 Mona AlQatari
A1 Sammy Stewart
A1 Ida Rishal
A1 Amelia Philpott
A1 Bernadett Kalmar
A1 Anna Kuta
A1 Michael Groves
A1 Nicholas Parkinson
A1 Abraham Acevedo-Arozena
A1 Sebastian Brandner
A1 David Bannerman
A1 Linda Greensmith
A1 Majid Hafezparast
A1 Martin Koltzenburg
A1 Robert Deacon
A1 Mike Fainzilber
A1 Elizabeth M. C. Fisher
YR 2011
UL http://www.jneurosci.org/content/31/14/5483.abstract
AB 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.