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- W2030247844 endingPage "113" @default.
- W2030247844 startingPage "91" @default.
- W2030247844 abstract "Animal models are pivotal for studies of pathogenesis and treatment of disorders of the central nervous system which in its complexity cannot yet be modeled in vitro or using computer simulations. The choice of a specific model to test novel therapeutic strategies for a human disease should be based on validity of the model for the approach: does the model reflect symptoms, pathogenesis and treatment response present in human patients? In the movement disorder dystonia, prior to the availability of genetically engineered mice, spontaneous mutants were chosen based on expression of dystonic features, including abnormal muscle contraction, movements and postures. Recent discovery of a number of genes and gene products involved in dystonia initiated research on pathogenesis of the disorder, and the creation of novel models based on gene mutations. Here we present a review of current models of dystonia, with a focus on genetic rodent models, which will likely be first choice in the future either for pathophysiological or for preclinical drug testing or both. In order to help selection of a model depending on expression of a specific feature of dystonia, this review is organized by symptoms and current knowledge of pathogenesis of dystonia. We conclude that albeit there is increasing need for research on pathogenesis of the disease and development of improved models, current models do replicate features of dystonia and are useful tools to develop urgently demanded treatment for this debilitating disorder." @default.
- W2030247844 created "2016-06-24" @default.
- W2030247844 creator A5058647332 @default.
- W2030247844 creator A5082199040 @default.
- W2030247844 date "2014-10-01" @default.
- W2030247844 modified "2023-10-10" @default.
- W2030247844 title "Genetic animal models of dystonia: Common features and diversities" @default.
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