ArticleAnnals of clinical and translational neurology2018
An image-based model of brain volume biomarker changes in Huntington's disease.
Article in Annals of clinical and translational neurology, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 31 papers.
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Who cites it
31 citing papers in PubMed, 59 citations in OpenAlex.
- Mapping brain volume changes in the zQ175DN mouse model of Huntington's disease: a longitudinal MRI study.Biomarker research · 2026Article
- Anterior Insula Drives Progressive Structural Brain Network Atrophy in the Behavioural Variant of Frontotemporal Dementia.Human brain mapping · 2025Article
- Staging of Alzheimer's disease progression in Down syndrome using mixed clinical and plasma biomarker measures with machine learning.Alzheimer's & dementia : the journal of the Alzheimer's Association · 2025Article
- A data-driven model of disability progression in progressive multiple sclerosis.Brain communications · 2025Article
- Identifying and verifying Huntington's disease subtypes: Clinical features, neuroimaging, and cytokine changes.Brain and behavior · 2024Article
- Data-driven modelling of neurodegenerative disease progression: thinking outside the black box.Nature reviews. Neuroscience · 2024Review
- Cerebrospinal Fluid Flow in Patients with Huntington's Disease.Annals of neurology · 2023Article
- Ex vivo 100 μm isotropic diffusion MRI-based tractography of connectivity changes in the end-stage R6/2 mouse model of Huntington's disease.Neuroprotection (Chichester, England) · 2023Article
- The temporal event-based model: Learning event timelines in progressive diseases.Imaging neuroscience (Cambridge, Mass.) · 2023Article
- Neurodegenerative disease of the brain: a survey of interdisciplinary approaches.Journal of the Royal Society, Interface · 2023Review
- Comparison and aggregation of event sequences across ten cohorts to describe the consensus biomarker evolution in Alzheimer's disease.Alzheimer's research & therapy · 2022Article
- Targeted Screening for Alzheimer's Disease Clinical Trials Using Data-Driven Disease Progression Models.Frontiers in artificial intelligence · 2022Article
- Temporal Progression Patterns of Brain Atrophy in Corticobasal Syndrome and Progressive Supranuclear Palsy Revealed by Subtype and Stage Inference (SuStaIn).Frontiers in neurology · 2022Article
- Preventive drugs for Huntington's disease: A choice-based conjoint survey of patient preferences.Journal of clinical and translational science · 2022Article
- A data-driven model of brain volume changes in progressive supranuclear palsy.Brain communications · 2022Article
- Brain Alterations in Aged OVT73 Sheep Model of Huntington's Disease: An MRI Based Approach.Journal of Huntington's disease · 2022Article
- Revealing the Timeline of Structural MRI Changes in Premanifest to Manifest Huntington Disease.Neurology. Genetics · 2021Article
- Sequence of clinical and neurodegeneration events in Parkinson's disease progression.Brain : a journal of neurology · 2021Observational
- Clinical, Imaging, and Laboratory Markers of Premanifest Spinocerebellar Ataxia 1, 2, 3, and 6: A Systematic Review.Journal of clinical neurology (Seoul, Korea) · 2021Review
- Inter-Cohort Validation of SuStaIn Model for Alzheimer's Disease.Frontiers in big data · 2021Article
Corrections and comments
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Authors and funding
11 authors at 2 institutions in 2 countries.
Funding
Abstract
objectiveDetermining the sequence in which Huntington's disease biomarkers become abnormal can provide important insights into the disease progression and a quantitative tool for patient stratification. Here, we construct and present a uniquely fine-grained model of temporal progression of Huntington's disease from premanifest through to manifest stages.
methodsWe employ a probabilistic event-based model to determine the sequence of appearance of atrophy in brain volumes, learned from structural MRI in the Track-HD study, as well as to estimate the uncertainty in the ordering. We use longitudinal and phenotypic data to demonstrate the utility of the patient staging system that the resulting model provides.
resultsThe model recovers the following order of detectable changes in brain region volumes: putamen, caudate, pallidum, insula white matter, nonventricular cerebrospinal fluid, amygdala, optic chiasm, third ventricle, posterior insula, and basal forebrain. This ordering is mostly preserved even under cross-validation of the uncertainty in the event sequence. Longitudinal analysis performed using 6 years of follow-up data from baseline confirms efficacy of the model, as subjects consistently move to later stages with time, and significant correlations are observed between the estimated stages and nonimaging phenotypic markers.
interpretationWe used a data-driven method to provide new insight into Huntington's disease progression as well as new power to stage and predict conversion. Our results highlight the potential of disease progression models, such as the event-based model, to provide new insight into Huntington's disease progression and to support fine-grained patient stratification for future precision medicine in Huntington's disease.
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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.