Dystonia: Are animal models relevant in therapeutics?

Revue neurologique
M MeringoloAntonio Pisani

Abstract

Dystonia refers to a heterogeneous group of movement disorders characterized by involuntary, sustained muscle contractions leading to repetitive twisting movements and abnormal postures. A better understanding of the etiology, pathogenesis and molecular mechanisms underlying dystonia may be obtained from animal models. Indeed, while studies in vitro using cell and tissue models are helpful for investigating molecular pathways, animal models remain essential for studying the pathogenesis of these disorders and exploring new potential treatment strategies. To date, the mouse is the most common choice for mammalian models in most laboratories, particularly when manipulations of the genome are planned. Dystonia animal models can be classified into two categories, etiological and symptomatic, although neither is able to recapitulate all features of these disorders in humans. Nevertheless, etiological and symptomatic animal models have advantages and limitations that should be taken into consideration according to the specific proposed hypothesis and experimental goals. Etiological mouse models of inherited dystonia can reproduce the etiology of the disorder and help to reveal biochemical and cellular alterations, although a large ma...Continue Reading

Citations

Jun 5, 2019·Expert Opinion on Drug Discovery·Sarah Pirio Richardson, H A Jinnah
Oct 30, 2019·Acta neurochirurgica·Ryoong Huh, Moonyoung Chung
Jan 19, 2021·Movement Disorders : Official Journal of the Movement Disorder Society·Chiara MelisMichelle E Ehrlich
Apr 15, 2020·Journal of Neuroscience Methods·P ImbrianiA Pisani
Jun 27, 2021·Movement Disorders : Official Journal of the Movement Disorder Society·Annalisa TassoneAntonio Pisani

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