1 40 1 Text A resource consisting primarily of words for reading. Examples include books, letters, dissertations, poems, newspapers, articles, archives of mailing lists. Note that facsimiles or images of texts are still of the genre Text. URL Address <a href="http://doi.org/10.1016/j.bbr.2018.01.029" target="_blank" rel="noreferrer noopener">http://doi.org/10.1016/j.bbr.2018.01.029</a> Pages 41–49 Volume 343 Dublin Core The Dublin Core metadata element set is common to all Omeka records, including items, files, and collections. For more information see, http://dublincore.org/documents/dces/. Title A name given to the resource Exacerbation of sensorimotor dysfunction in mice deficient in Atp13a2 and overexpressing human wildtype alpha-synuclein. Publisher An entity responsible for making the resource available Behavioural brain research Date A point or period of time associated with an event in the lifecycle of the resource 2018 2018-05 Subject The topic of the resource *Alpha-synuclein; *ATP13A2; *Mice; *Phenotype; *Sensorimotor; Adenosine Triphosphatases/*deficiency/genetics; alpha-Synuclein/genetics/*metabolism; Animal; Animals; Body Temperature; Body Weight; Disease Models; Female; Gait Disorders; Humans; Inbred C57BL; Male; Membrane Proteins/*deficiency/genetics; Mice; Motor Skills/physiology; Neurologic/*metabolism; Phenotype; Severity of Illness Index; Sex Characteristics; Transgenic Creator An entity primarily responsible for making the resource Dirr Emily R; Ekhator Osunde R; Blackwood Rachel; Holden John G; Masliah Eliezer; Schultheis Patrick J; Fleming Sheila M Description An account of the resource Loss of function mutations in the gene ATP13A2 are associated with Kufor-Rakeb Syndrome and Neuronal Ceroid Lipofuscinosis, the former designated as an inherited form of Parkinson's disease (PD). The function of ATP13A2 is unclear but in vitro studies indicate it is a lysosomal protein and may interact with the presynaptic protein alpha-synuclein (aSyn) and certain heavy metals. Accumulation of aSyn is a major component of lewy bodies, the pathological hallmark of PD. Atp13a2-deficient (13a2) mice develop age-dependent sensorimotor deficits, and accumulation of insoluble aSyn in the brain. To better understand the interaction between ATP13A2 and aSyn, double mutant mice with loss of Atp13a2 function combined with overexpression of human wildtype aSyn were generated. Female and male wildtype (WT), 13a2, aSyn, and 13a2-aSyn mice were tested on a battery of sensorimotor tests including adhesive removal, challenging beam traversal, spontaneous activity, gait, locomotor activity, and nest-building at 2, 4, and 6 months of age. Double mutant mice showed an earlier onset and accelerated alterations in sensorimotor function that were age, sex and test-dependent. Female 13a2-aSyn mice showed early and progressive dysfunction on the beam and in locomotor activity. In males, 13a2-aSyn mice showed more severe impairments in spontaneous activity and adhesive removal. Sex differences were also observed in aSyn and 13a2-aSyn mice on the beam, cylinder, and adhesive removal tests. In other tasks, double mutant mice displayed deficits similar to aSyn mice. These results indicate loss of Atp13a2 function exacerbates the sensorimotor phenotype in aSyn mice in an age and sex-dependent manner. Identifier An unambiguous reference to the resource within a given context <a href="http://doi.org/10.1016/j.bbr.2018.01.029" target="_blank" rel="noreferrer noopener">10.1016/j.bbr.2018.01.029</a> Rights Information about rights held in and over the resource Article information provided for research and reference use only. All rights are retained by the journal listed under publisher and/or the creator(s). *Alpha-synuclein *ATP13A2 *Mice *Phenotype *Sensorimotor 2018 Adenosine Triphosphatases/*deficiency/genetics alpha-Synuclein/genetics/*metabolism Animal Animals Behavioural brain research Blackwood Rachel Body Temperature Body Weight Department of Pharmaceutical Sciences Dirr Emily R Disease Models Ekhator Osunde R Female Fleming Sheila M Gait Disorders Holden John G Humans Inbred C57BL Male Masliah Eliezer Membrane Proteins/*deficiency/genetics Mice Motor Skills/physiology NEOMED College of Pharmacy Neurologic/*metabolism Phenotype Schultheis Patrick J Severity of Illness Index Sex Characteristics Transgenic