Publication Date

2014

Document Type

Thesis

Committee Members

Mark Rich, M.D., Ph.D. (Committee Chair); Sheila Fleming, Ph.D. (Committee Member); John Holden, Ph.D. (Committee Member)

Degree Name

Master of Science (MS)

Abstract

Abnormal accumulation of the presynaptic protein alpha-synuclein (aSyn) is associated with both familial and sporadic forms of Parkinson's disease. ATP13A2 is a lysosomal protein involved in protein degradation, including aSyn. Mice with a loss of ATP13A2 function develop age-related sensorimotor impairments and, in the brain, enhanced lipofuscin and insoluble aSyn accumulation. To further examine the potential interaction between aSyn and ATP13A2, aSyn overexpressing mice (aSyn) were crossbred with Atp13a2-defeicient (13a2) mice. Wildtype (WT), 13a2, aSyn, and double mutant aSyn/13a2 mice were generated and tested behaviorally at 3 ages. Sensorimotor function and cognition were examined using a battery of behavioral tests. Analysis of males and females revealed aSyn/13a2s make more errors on the beam, show increased hyperactivity and impulsivity, and build smaller nests compared to WTs. Female aSyn/13a2s display an earlier and progressive phenotype, suggesting increased aSyn burden combined with loss of ATP13A2 function alters sensorimotor function and cognition in mice.

Page Count

164

Department or Program

Department of Neuroscience, Cell Biology and Physiology

Year Degree Awarded

2014


Included in

Anatomy Commons

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