Literature DB >> 17532061

Behavioural characterisation of the robotic mouse mutant.

Peter L Oliver1, David A Keays, Kay E Davies.   

Abstract

The ataxic mouse mutant robotic is characterised by progressive adult-onset Purkinje cell loss that occurs in a distinctive region-specific pattern. We report the first behavioural characterisation of this mutant and quantify its performance on tests of motor function, locomotor and exploratory activity over a time course that reflects specific stages of cell loss in the cerebellum. Robotic mutants are significantly impaired on the rotarod and static rod tests of coordination and their performance declined during aging. In addition, gait analysis revealed an increase in the severity of the ataxia displayed by mutants over time. Interestingly, spontaneous alternation testing in a T-maze was not significantly affected in robotic mice, unlike other ataxic mutants with more rapid and extensive cerebellar degeneration; robotic therefore provides an opportunity to investigate the necessity of specific Purkinje cell populations for various behavioural tasks.

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Year:  2007        PMID: 17532061     DOI: 10.1016/j.bbr.2007.04.012

Source DB:  PubMed          Journal:  Behav Brain Res        ISSN: 0166-4328            Impact factor:   3.332


  11 in total

1.  Mutations in Vps15 perturb neuronal migration in mice and are associated with neurodevelopmental disease in humans.

Authors:  Thomas Gstrein; Andrew Edwards; Anna Přistoupilová; Ines Leca; Martin Breuss; Sandra Pilat-Carotta; Andi H Hansen; Ratna Tripathy; Anna K Traunbauer; Tobias Hochstoeger; Gavril Rosoklija; Marco Repic; Lukas Landler; Viktor Stránecký; Gerhard Dürnberger; Thomas M Keane; Johannes Zuber; David J Adams; Jonathan Flint; Tomas Honzik; Marta Gut; Sergi Beltran; Karl Mechtler; Elliott Sherr; Stanislav Kmoch; Ivo Gut; David A Keays
Journal:  Nat Neurosci       Date:  2018-01-08       Impact factor: 24.884

2.  Animal models of human cerebellar ataxias: a cornerstone for the therapies of the twenty-first century.

Authors:  Mario Manto; Daniele Marmolino
Journal:  Cerebellum       Date:  2009-09       Impact factor: 3.847

3.  The expression of TAG-1 in glial cells is sufficient for the formation of the juxtaparanodal complex and the phenotypic rescue of tag-1 homozygous mutants in the CNS.

Authors:  Maria Savvaki; Kostas Theodorakis; Lida Zoupi; Antonis Stamatakis; Simona Tivodar; Kyriacos Kyriacou; Fotini Stylianopoulou; Domna Karagogeos
Journal:  J Neurosci       Date:  2010-10-20       Impact factor: 6.167

4.  Smad2 protein disruption in the central nervous system leads to aberrant cerebellar development and early postnatal ataxia in mice.

Authors:  Lixiang Wang; Masatoshi Nomura; Yutaka Goto; Kimitaka Tanaka; Ryuichi Sakamoto; Ichiro Abe; Shohei Sakamoto; Atsushi Shibata; Patricio L M Enciso; Masahiro Adachi; Keizo Ohnaka; Hisaya Kawate; Ryoichi Takayanagi
Journal:  J Biol Chem       Date:  2011-04-04       Impact factor: 5.157

5.  Regulator of G protein signaling 6 (RGS6) protein ensures coordination of motor movement by modulating GABAB receptor signaling.

Authors:  Biswanath Maity; Adele Stewart; Jianqi Yang; Lipin Loo; David Sheff; Andrew J Shepherd; Durga P Mohapatra; Rory A Fisher
Journal:  J Biol Chem       Date:  2011-12-16       Impact factor: 5.157

6.  Prevention of dystrophic pathology in severely affected dystrophin/utrophin-deficient mice by morpholino-oligomer-mediated exon-skipping.

Authors:  Aurélie Goyenvalle; Arran Babbs; Dave Powell; Ryszard Kole; Sue Fletcher; Steve D Wilton; Kay E Davies
Journal:  Mol Ther       Date:  2009-10-20       Impact factor: 11.454

Review 7.  The robotic mouse: understanding the role of AF4, a cofactor of transcriptional elongation and chromatin remodelling, in purkinje cell function.

Authors:  Emmanuelle Bitoun; Kay E Davies
Journal:  Cerebellum       Date:  2009-04-02       Impact factor: 3.847

8.  Rescue of severely affected dystrophin/utrophin-deficient mice through scAAV-U7snRNA-mediated exon skipping.

Authors:  Aurélie Goyenvalle; Arran Babbs; Jordan Wright; Vivienne Wilkins; Dave Powell; Luis Garcia; Kay E Davies
Journal:  Hum Mol Genet       Date:  2012-03-02       Impact factor: 6.150

9.  A point mutation in TRPC3 causes abnormal Purkinje cell development and cerebellar ataxia in moonwalker mice.

Authors:  Esther B E Becker; Peter L Oliver; Maike D Glitsch; Gareth T Banks; Francesca Achilli; Andrea Hardy; Patrick M Nolan; Elizabeth M C Fisher; Kay E Davies
Journal:  Proc Natl Acad Sci U S A       Date:  2009-04-07       Impact factor: 11.205

10.  AF4 is a critical regulator of the IGF-1 signaling pathway during Purkinje cell development.

Authors:  Emmanuelle Bitoun; Mattéa J Finelli; Peter L Oliver; Sheena Lee; Kay E Davies
Journal:  J Neurosci       Date:  2009-12-09       Impact factor: 6.167

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