Title: Poster session: Alterations in gait, operant responding, and anxiety-related behaviour in a murine model of metachromatic leukodystrophy
Authors: Stroobants, Stijn
Leroy, Toon
Eckhardt, M.
Aerts, Jean-Marie
Berckmans, Daniel
D'Hooge, Rudi #
Issue Date: Jul-2010
Host Document: FENS abstract vol:5
Conference: FENS Formum of European Neuroscience edition:7 location:Amsterdam date:3-7 July 2010
Article number: 165.20
Abstract: Arylsulfatase A (ASA) -deficient mice represent an animal model for the lysosomal storage disorder metachromatic leukodystrophy (MLD). Although the model has been applied in pathophysiological and therapeutic studies, the behavioural phenotype of ASA-/- mice is only partially characterised, and the most decisive outcome measures for therapy evaluation only emerge beyond one year of age. Presently, ASA-/- mice and ASA+/- control mice were studied at six and twelve months of age on an extensive battery including tests of neuromotor ability, exploratory behaviour, and learning and memory. Overt signs of ataxia were not observed in six-month-old ASA-/- mice, but quantitative gait analysis during open field exploration revealed that ASA-/- mice displayed increased hind base width and increased stride lengths for all paws. Their covert motor incoordination was evident in a correlation analysis which unveiled decreased harmonisation of concurrent gait parameters. For example, while ASA+/- controls demonstrated substantial convergence of front and hind base width (r = 0.54), these variables actually diverged in ASA-/- mice (r = -0.37). Furthermore, various behavioural observations indicated emotional alterations in ASA-/- mice. Six-month-old ASA-/- mice also showed decreased response rates in scheduled operant responding. The present findings could provide relevant behavioural outcome measures for further use of this murine MLD model in preclinical studies
Publication status: published
KU Leuven publication type: IMa
Appears in Collections:Laboratory for Biological Psychology
Division M3-BIORES: Measure, Model & Manage Bioresponses (-)
# (joint) last author

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