PT  - JOURNAL ARTICLE
AU  - Gareth T. Banks
AU  - Matilda A. Haas
AU  - Samantha Line
AU  - Hazel L. Shepherd
AU  - Mona AlQatari
AU  - Sammy Stewart
AU  - Ida Rishal
AU  - Amelia Philpott
AU  - Bernadett Kalmar
AU  - Anna Kuta
AU  - Michael Groves
AU  - Nicholas Parkinson
AU  - Abraham Acevedo-Arozena
AU  - Sebastian Brandner
AU  - David Bannerman
AU  - Linda Greensmith
AU  - Majid Hafezparast
AU  - Martin Koltzenburg
AU  - Robert Deacon
AU  - Mike Fainzilber
AU  - Elizabeth M. C. Fisher
TI  - Behavioral and Other Phenotypes in a Cytoplasmic Dynein Light Intermediate Chain 1 Mutant Mouse
AID  - 10.1523/JNEUROSCI.5244-10.2011
DP  - 2011 Apr 06
TA  - The Journal of Neuroscience
PG  - 5483--5494
VI  - 31
IP  - 14
4099  - http://www.jneurosci.org/content/31/14/5483.short
4100  - http://www.jneurosci.org/content/31/14/5483.full
SO  - J. Neurosci.2011 Apr 06; 31
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.