ArticleNeurology2020
Abnormal development of cerebellar-striatal circuitry in Huntington disease.
Article in Neurology, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers, 1 of them a synthesis that pooled it.
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Who cites it
43 citing papers in PubMed, 1 synthesis or guideline pooled it, 66 citations in OpenAlex.
- The Role of the Cerebellum in Huntington's Disease: a Systematic Review.Cerebellum (London, England) · 2021Pooled it
- Genetic frontotemporal degeneration across the lifespan? A critical appraisal of the neurodevelopmental hypothesis.Alzheimer's & dementia : the journal of the Alzheimer's Association · 2026Review
- Insights into neurodevelopmental features of Huntington's disease from stem cell-derived models including organoids.Journal of Huntington's disease · 2026Review
- Observational
- Preoperative Functional Connectivity Predicts Antiparkinson Drug Change after Deep Brain Stimulation.Movement disorders : official journal of the Movement Disorder Society · 2026Article
- Neurodevelopmental effects of genetic frontotemporal dementia mutations revealed by total intracranial volume differences.Journal of Alzheimer's disease : JAD · 2026Article
- Cerebellar motor and non-motor contributions to dystonia pathophysiology and treatment.Dystonia (Lausanne, Switzerland) · 2026Article
- Distinct molecular patterns in R6/2 HD mouse brain: Insights from spatiotemporal transcriptomics.Neuron · 2025Article
- Along-Tract Diffusion Alterations in the Dentato-Rubro-Thalamic Tract Correlate With Motor and Cognitive Decline in Huntington's Disease.Human brain mapping · 2025Article
- Musical Distractions: Music-Based Rhythmic Auditory Stimulation Fails to Improve Gait in Huntington's Disease.Brain sciences · 2025Article
- Cell-specific mechanisms drive connectivity across the time course of Huntington's disease.Nature communications · 2025Article
- Cortical and Striatal Functional Connectivity in Juvenile-Onset Huntington's Disease.Brain sciences · 2025Article
- Mutant Huntingtin Drives Development of an Advantageous Brain Early in Life: Evidence in Support of Antagonistic Pleiotropy.Annals of neurology · 2024Article
- Dendrite injury triggers neuroprotection in Drosophila models of neurodegenerative disease.Scientific reports · 2024Article
- Evolution of aberrant brain-wide spatiotemporal dynamics of resting-state networks in a Huntington's disease mouse model.Clinical and translational medicine · 2024Article
- Altered Iron and Microstructure in Huntington's Disease Subcortical Nuclei: Insight From 7T MRI.Journal of magnetic resonance imaging : JMRI · 2024Article
- Systems Genetics Analyses Reveals Key Genes Related to Behavioral Traits in the Striatum of CFW Mice.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2024Article
- Review
- Review
- Impairments to executive function in emerging adults with Huntington disease.Journal of neurology, neurosurgery, and psychiatry · 2023Article
Corrections and comments
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Authors and funding
6 authors at 1 institution in 1 country.
Funding
Abstract
objectiveTo test the hypothesis that the trajectory of functional connections over time of the striatum and the cerebellum differs between presymptomatic patients with the Huntington disease (HD) gene expansion (GE) and patients with a family history of HD but without the GE (GNE), we evaluated functional MRI data from the Kids-HD study.
methodsWe utilized resting-state, functional MRI data from participants in the Kids-HD study between 6 and 18 years old. Participants were divided into GE (CAG 36-59) and GNE (CAG <36) groups. Seed-to-seed correlations were calculated among 4 regions that provide input signals to the anterior cerebellum: (1) dorsocaudal putamen, (2) globus pallidus externa, (3) subthalamic nucleus, and (4) pontine nuclei; and 2 regions that represented output from the cerebellum: the dentate nucleus to the (1) ventrolateral thalamus and (2) dorsocaudal putamen. Linear mixed effects regression models evaluated differences in developmental trajectories of these connections over time between groups.
resultsFour of the six striatal-cerebellum correlations showed significantly different trajectories between groups. All showed a pattern where in the early age ranges (6-12 years) there was hyperconnectivity in the GE compared to the GNE, with those trajectories showing linear decline in the latter half of the age range.
conclusionThese results parallel previous findings showing striatal hypertrophy in children with GE as early as age 6. These findings support the notion of developmentally higher connectivity between the striatum and cerebellum early in the life of the child with HD GE, possibly setting the stage for cerebellar compensatory mechanisms.
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Registered trials
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