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- W3132734562 endingPage "421" @default.
- W3132734562 startingPage "421" @default.
- W3132734562 abstract "Hereditary dominant ataxias are a heterogeneous group of neurodegenerative conditions causing cerebellar dysfunction and characterized by progressive motor incoordination. Despite many efforts put into the study of these diseases, there are no effective treatments yet. Zebrafish models are widely used to characterize neuronal disorders due to its conserved vertebrate genetics that easily support genetic edition and their optic transparency that allows observing the intact CNS and its connections. In addition, its small size and external fertilization help to develop high throughput assays of candidate drugs. Here, we discuss the contributions of zebrafish models to the study of dominant ataxias defining phenotypes, genetic function, behavior and possible treatments. In addition, we review the zebrafish models created for X-linked repeat expansion diseases X-fragile/fragile-X tremor ataxia. Most of the models reviewed here presented neuronal damage and locomotor deficits. However, there is a generalized lack of zebrafish adult heterozygous models and there are no knock-in zebrafish models available for these diseases. The models created for dominant ataxias helped to elucidate gene function and mechanisms that cause neuronal damage. In the future, the application of new genetic edition techniques would help to develop more accurate zebrafish models of dominant ataxias." @default.
- W3132734562 created "2021-03-01" @default.
- W3132734562 creator A5019649839 @default.
- W3132734562 creator A5020653832 @default.
- W3132734562 creator A5057384646 @default.
- W3132734562 creator A5057447134 @default.
- W3132734562 creator A5080569326 @default.
- W3132734562 date "2021-02-17" @default.
- W3132734562 modified "2023-10-10" @default.
- W3132734562 title "Zebrafish Models of Autosomal Dominant Ataxias" @default.
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