ArticleScientific reports2019
Cortical circuit alterations precede motor impairments in Huntington's disease mice.
Article in Scientific reports, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.
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33 citing papers in PubMed, 70 citations in OpenAlex.
- Myelin Dynamics at the Axon-Oligodendrocyte Interface: Adaptive Conduction Homeostasis in Demyelination, Remyelination and White Matter Repair.International journal of molecular sciences · 2026Review
- Article
- Anle138b ameliorates pathological phenotypes in mouse and cellular models of Huntington's disease.EMBO molecular medicine · 2026Article
- Human stem cell-derived neurons establish functional inhibitory-excitatory cortical circuits in a chimeric transplantation model.Scientific reports · 2026Article
- Presymptomatic targeted circuit manipulation for ameliorating Huntington's disease pathogenesis.iScience · 2025Article
- Optogenetic restoration of neuron subtype-specific cortical activity ameliorates motor deficits in Huntington's Disease mice.bioRxiv : the preprint server for biology · 2025Article
- Sleep disturbance in clinical and preclinical scrapie-infected sheep measured by polysomnography.The veterinary quarterly · 2024Article
- Presymptomatic Targeted Circuit Manipulation for Ameliorating Huntington's Disease Pathogenesis.bioRxiv : the preprint server for biology · 2024Article
- Motor skill learning modulates striatal extracellular vesicles' content in a mouse model of Huntington's disease.Cell communication and signaling : CCS · 2024Article
- Longitudinal alterations in brain perfusion and vascular reactivity in the zQ175DN mouse model of Huntington's disease.Journal of biomedical science · 2024Article
- Microglia and complement mediate early corticostriatal synapse loss and cognitive dysfunction in Huntington's disease.Nature medicine · 2023Article
- Neuroprotective effects of hepatoma-derived growth factor in models of Huntington's disease.Life science alliance · 2023Article
- Article
- Motor Imagery Analysis from Extensive EEG Data Representations Using Convolutional Neural Networks.Sensors (Basel, Switzerland) · 2022Article
- Synaptic Dysfunction in Huntington's Disease: Lessons from Genetic Animal Models.The Neuroscientist : a review journal bringing neurobiology, neurology and psychiatry · 2022Article
- Calcium imaging: A versatile tool to examine Huntington's disease mechanisms and progression.Frontiers in neuroscience · 2022Review
- Distinct histological alterations of cortical interneuron types in mouse models of Huntington's disease.Frontiers in neuroscience · 2022Article
- Long-term dynamics of aberrant neuronal activity in awake Alzheimer's disease transgenic mice.Communications biology · 2021Article
- Non-Cell Autonomous and Epigenetic Mechanisms of Huntington's Disease.International journal of molecular sciences · 2021Review
- Fluc-EGFP reporter mice reveal differential alterations of neuronal proteostasis in aging and disease.The EMBO journal · 2021Article
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Authors and funding
11 authors at 4 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Huntington's disease (HD) is a devastating hereditary movement disorder, characterized by degeneration of neurons in the striatum and cortex. Studies in human patients and mouse HD models suggest that disturbances of neuronal function in the neocortex play an important role in disease onset and progression. However, the precise nature and time course of cortical alterations in HD have remained elusive. Here, we use chronic in vivo two-photon calcium imaging to longitudinally monitor the activity of identified single neurons in layer 2/3 of the primary motor cortex in awake, behaving R6/2 transgenic HD mice and wildtype littermates. R6/2 mice show age-dependent changes in cortical network function, with an increase in activity that affects a large fraction of cells and occurs rather abruptly within one week, preceeding the onset of motor defects. Furthermore, quantitative proteomics demonstrate a pronounced downregulation of synaptic proteins in the cortex, and histological analyses in R6/2 mice and human HD autopsy cases reveal a reduction in perisomatic inhibitory synaptic contacts on layer 2/3 pyramidal cells. Taken together, our study provides a time-resolved description of cortical network dysfunction in behaving HD mice and points to disturbed excitation/inhibition balance as an important pathomechanism in HD.
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