ArticleNeurobiology of disease2016
In vivo imaging reveals impaired connectivity across cortical and subcortical networks in a mouse model of DYT1 dystonia.
Article in Neurobiology of disease, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers, 1 of them a synthesis that pooled it.
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Who cites it
19 citing papers in PubMed, 1 synthesis or guideline pooled it, 31 citations in OpenAlex.
- Structural magnetic resonance imaging in dystonia: A systematic review of methodological approaches and findings.European journal of neurology · 2022Pooled it
- Deutetrabenazine in Hyperkinetic Movement Disorders: VMAT2 Modulation From Choreiform Disorders to Hyperkinetic Dystonic Syndromes.Journal of central nervous system disease · 2026Review
- Motor and sensory neurophysiology in relation to [Brain communications · 2025Article
- Altered Functional Brain Connectivity in Dyt1 Knock-in mouse models.Dystonia (Lausanne, Switzerland) · 2025Article
- Cell-specific Dyt1 ∆GAG knock-in to basal ganglia and cerebellum reveal differential effects on motor behavior and sensorimotor network function.Experimental neurology · 2023Article
- Evaluation of an Adoptive Cellular Therapy-Based Vaccine in a Transgenic Mouse Model of α-synucleinopathy.ACS chemical neuroscience · 2023Article
- DYT-Frontiers in neuroscience · 2023Review
- Dystonia in Childhood: How Insights from Paediatric Research Enrich the Network Theory of Dystonia.Advances in neurobiology · 2023Article
- The HIV protease inhibitor, ritonavir, corrects diverse brain phenotypes across development in mouse model of DYT-TOR1A dystonia.Science translational medicine · 2021Article
- Functional connectivity of the motor system in dystonia due to PKAN.eNeurologicalSci · 2021Article
- Sensorimotor Integration in Childhood Dystonia and Dystonic Cerebral Palsy-A Developmental Perspective.Frontiers in neurology · 2021Review
- The abnormal firing of Purkinje cells in the knockin mouse model of DYT1 dystonia.Brain research bulletin · 2020Article
- Abnormal microscale neuronal connectivity triggered by a proprioceptive stimulus in dystonia.Scientific reports · 2020Article
- Common functional networks in the mouse brain revealed by multi-centre resting-state fMRI analysis.NeuroImage · 2020Article
- Diffusion magnetic resonance imaging-derived free water detects neurodegenerative pattern induced by interferon-γ.Brain structure & function · 2020Article
- Modern approaches for modelling dystonia and Huntington's disease in vitro and in vivo.International journal of experimental pathology · 2019Review
- The basal ganglia and the cerebellum: nodes in an integrated network.Nature reviews. Neuroscience · 2018Review
- Forebrain knock-out of torsinA reduces striatal free-water and impairs whole-brain functional connectivity in a symptomatic mouse model of DYT1 dystonia.Neurobiology of disease · 2017Article
- The Anatomical Basis for Dystonia: The Motor Network Model.Tremor and other hyperkinetic movements (New York, N.Y.) · 2017Review
Corrections and comments
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Authors and funding
7 authors at 1 institution in 1 country.
Funding
Abstract
Developing in vivo functional and structural neuroimaging assays in Dyt1 ΔGAG heterozygous knock-in (Dyt1 KI) mice provide insight into the pathophysiology underlying DYT1 dystonia. In the current study, we examined in vivo functional connectivity of large-scale cortical and subcortical networks in Dyt1 KI mice and wild-type (WT) controls using resting-state functional magnetic resonance imaging (MRI) and an independent component analysis. In addition, using diffusion MRI we examined how structural integrity across the basal ganglia and cerebellum directly relates to impairments in functional connectivity. Compared to WT mice, Dyt1 KI mice revealed increased functional connectivity across the striatum, thalamus, and somatosensory cortex; and reduced functional connectivity in the motor and cerebellar cortices. Further, Dyt1 KI mice demonstrated elevated free-water (FW) in the striatum and cerebellum compared to WT mice, and increased FW was correlated with impairments in functional connectivity across basal ganglia, cerebellum, and sensorimotor cortex. The current study provides the first in vivo MRI-based evidence in support of the hypothesis that the deletion of a 3-base pair (ΔGAG) sequence in the Dyt1 gene encoding torsinA has network level effects on in vivo functional connectivity and microstructural integrity across the sensorimotor cortex, basal ganglia, and cerebellum.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.