ArticleMovement disorders : official journal of the Movement Disorder Society2017
Parkinsonism without dopamine neuron degeneration in aged l-dopa-responsive dystonia knockin mice.
Article in Movement disorders : official journal of the Movement Disorder Society, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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9 citing papers in PubMed, 15 citations in OpenAlex.
- Tyrosine Hydroxylase Deficiency Impairs TH Axonal Transport, Brain Function, and Neuronal Plasticity.Journal of inherited metabolic disease · 2026Article
- Striatal cell-type-specific molecular signatures reveal potential therapeutic targets in a model of dystonia.Neurobiology of disease · 2025Article
- Clinical and Basic Research on Dopa-Responsive Dystonia: Neuropathological and Neurochemical Findings.Juntendo medical journal · 2025Article
- Synaptic Dysfunction in Dystonia: Update From Experimental Models.Current neuropharmacology · 2023Review
- Exploring the link between Parkinson's disease and type 2 diabetes mellitus in Drosophila.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2022Article
- Differential expression of striatal proteins in a mouse model of DOPA-responsive dystonia reveals shared mechanisms among dystonic disorders.Molecular genetics and metabolism · 2021Article
- GCH-1 genetic variant may cause Parkinsonism by unmasking the subclinical nigral pathology.Journal of neurology · 2020Article
- Roles of the MMovement disorders : official journal of the Movement Disorder Society · 2019Review
- Striatal Cholinergic Interneurons in a Knock-in Mouse Model of L-DOPA-Responsive Dystonia.Frontiers in systems neuroscience · 2018Article
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8 authors at 1 institution in 1 country.
Funding
Abstract
backgroundRecent neuroimaging studies implicate nigrostriatal degeneration as a critical factor in producing late-onset parkinsonism in patients with l-dopa-responsive dystonia-causing mutations. However, postmortem anatomical studies do not reveal neurodegeneration in l-dopa-responsive dystonia patients. These contrasting findings make it unclear how parkinsonism develops in l-dopa-responsive dystonia mutation carriers.
methodsWe prospectively assessed motor dysfunction, responses to dopaminergic challenge, and dopamine neuron degeneration with aging in a validated knockin mouse model bearing a l-dopa-responsive dystonia-causing mutation found in humans.
resultsAs l-dopa-responsive dystonia mice aged, dystonic movements waned while locomotor activity decreased and initiation of movements slowed. Despite the age-related reduction in movement, there was no evidence for degeneration of midbrain dopamine neurons. Presynaptically mediated dopaminergic responses did not change with age in l-dopa-responsive dystonia mice, but responses to D1 dopamine receptor agonists decreased with age.
conclusionsWe have demonstrated for the first time the co-occurrence of dystonia and Parkinson's-like features (mainly consisting of hypokinesia) in a genetic mouse model. In this model we show that these features evolve without dopaminergic neurodegeneration, suggesting that postsynaptic plasticity, rather than presynaptic degeneration, may contribute to the development of parkinsonism in patients with l-dopa-responsive dystonia. © 2017 International Parkinson and Movement Disorder Society.
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