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• W2017604310 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 characterized, and the most decisive outcome measures for therapy evaluation only emerge beyond 1 year of age. Presently, ASA−/− mice and ASA+/− control mice were studied at 6 and 12 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 6-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." @default.
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• W2017604310 date "2008-06-01" @default.
• W2017604310 modified "2023-10-16" @default.
• W2017604310 title "Early signs of neurolipidosis-related behavioural alterations in a murine model of metachromatic leukodystrophy" @default.
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• W2017604310 doi "https://doi.org/10.1016/j.bbr.2008.01.008" @default.
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