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dc.contributor.authorConnell, James W.
dc.contributor.authorAllison, Rachel
dc.contributor.authorReid, Evan
dc.date.accessioned2016-08-01T15:41:48Z
dc.date.available2016-08-01T15:41:48Z
dc.date.issued2016-03-28
dc.identifier.citationConnell et al. PLOS ONE (2016) Vol. 11, Issue 3, Article e0152413. doi:10.1371/ journal.pone.0152413en_US
dc.identifier.urihttps://www.repository.cam.ac.uk/handle/1810/256934
dc.description.abstractThe hereditary spastic paraplegias (HSPs) are genetic conditions in which there is progressive axonal degeneration in the corticospinal tract. Autosomal dominant mutations, including nonsense, frameshift and missense changes, in the gene encoding the microtubule severing ATPase spastin are the most common cause of HSP in North America and northern Europe. In this study we report quantitative gait analysis using a motorized treadmill system, carried out on mice knocked-in for a disease-associated mutation affecting a critical residue in the Walker A motif of the spastin ATPase domain. At 4 months and at one year of age homozygous mutant mice had a number of abnormal gait parameters, including in stride length and stride duration, compared to heterozygous and wild-type littermates. Gait parameters in heterozygous animals did not differ from wild-type littermates. We conclude that quantitative gait analysis using the DigiGait system sensitively detects motor abnormalities in a hereditary spastic paraplegia model, and would be a useful method for analyzing the effects of pharmacological treatments for HSP.en
dc.language.isoenen
dc.publisherPLOSen
dc.rightsAll Rights Reserveden
dc.rights.urihttps://www.rioxx.net/licenses/all-rights-reserved/en
dc.titleQuantitative Gait Analysis Using a Motorized Treadmill System Sensitively Detects Motor Abnormalities in Mice Expressing ATPase Defective Spastinen
dc.typeArticleen
dc.date.updated2016-07-05T10:08:44Z
dc.identifier.doi10.17863/CAM.543
dcterms.dateAccepted2016
rioxxterms.versionVoRen


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