ArticleMovement disorders : official journal of the Movement Disorder Society2015
Multivariate prediction of motor diagnosis in Huntington's disease: 12 years of PREDICT-HD.
Article in Movement disorders : official journal of the Movement Disorder Society, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers.
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Who cites it
28 citing papers in PubMed, 44 citations in OpenAlex.
- Sample enrichment for clinical trials to show delay of onset in huntington disease.Movement disorders : official journal of the Movement Disorder Society · 2019Trial
- Discovery and Targeted Proteomic Studies Reveal Striatal Markers Validated for Huntington's Disease.Annals of clinical and translational neurology · 2026Article
- Observational
- Beyond Alzheimer's disease-translating biomarker insights across CNS diseases.Science translational medicine · 2025Review
- Childhood to Adult Neurodevelopment in Gene-Expanded Huntington's Disease (ChANGE-HD) study protocol: A Prospective Longitudinal Neurodevelopmental Study of Huntington's Disease.medRxiv : the preprint server for health sciences · 2025Article
- clusterMLD: An Efficient Hierarchical Clustering Method for Multivariate Longitudinal Data.Journal of computational and graphical statistics : a joint publication of American Statistical Association, Institute of Mathematical Statistics, Interface Foundation of North America · 2023Article
- Forecasting individual progression trajectories in Huntington disease enables more powered clinical trials.Scientific reports · 2022Article
- Estimating disease onset from change points of markers measured with error.Biostatistics (Oxford, England) · 2021Observational
- Longitudinal subcortical segmentation with deep learning.Proceedings of SPIE--the International Society for Optical Engineering · 2021Article
- MRI subcortical segmentation in neurodegeneration with cascaded 3D CNNs.Proceedings of SPIE--the International Society for Optical Engineering · 2021Article
- Disease-related Huntingtin seeding activities in cerebrospinal fluids of Huntington's disease patients.Scientific reports · 2020Article
- Generalizing MRI Subcortical Segmentation to Neurodegeneration.Machine learning in clinical neuroimaging and radiogenomics in neuro-oncology : Third International Workshop, MLCN 2020, and Second International Workshop, RNO-AI 2020, held in conjunction with MICCAI 2020, Lima, Peru, October 4-8, 2020... · 2020Article
- Huntington disease: Advances in the understanding of its mechanisms.Clinical parkinsonism & related disorders · 2020Review
- Predicting the Risk of Huntington's Disease with Multiple Longitudinal Biomarkers.Journal of Huntington's disease · 2019Observational
- Joint modeling of multivariate longitudinal data and survival data in several observational studies of Huntington's disease.BMC medical research methodology · 2018Article
- Genetic Modification of Huntington Disease Acts Early in the Prediagnosis Phase.American journal of human genetics · 2018Article
- High and Low Levels of an NTRK2-Driven Genetic Profile Affect Motor- and Cognition-Associated Frontal Gray Matter in Prodromal Huntington's Disease.Brain sciences · 2018Article
- Translation of MicroRNA-Based Huntingtin-Lowering Therapies from Preclinical Studies to the Clinic.Molecular therapy : the journal of the American Society of Gene Therapy · 2018Review
- Genetics Modulate Gray Matter Variation Beyond Disease Burden in Prodromal Huntington's Disease.Frontiers in neurology · 2018Article
- Baseline multimodal information predicts future motor impairment in premanifest Huntington's disease.NeuroImage. Clinical · 2018Article
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
backgroundIt is well known in Huntington's disease that cytosine-adenine-guanine expansion and age at study entry are predictive of the timing of motor diagnosis. The goal of this study was to assess whether additional motor, imaging, cognitive, functional, psychiatric, and demographic variables measured at study entry increased the ability to predict the risk of motor diagnosis over 12 years.
methodsOne thousand seventy-eight Huntington's disease gene-expanded carriers (64% female) from the Neurobiological Predictors of Huntington's Disease study were followed up for up to 12 y (mean = 5, standard deviation = 3.3) covering 2002 to 2014. No one had a motor diagnosis at study entry, but 225 (21%) carriers prospectively received a motor diagnosis. Analysis was performed with random survival forests, which is a machine learning method for right-censored data.
resultsAdding 34 variables along with cytosine-adenine-guanine and age substantially increased predictive accuracy relative to cytosine-adenine-guanine and age alone. Adding six of the common motor and cognitive variables (total motor score, diagnostic confidence level, Symbol Digit Modalities Test, three Stroop tests) resulted in lower predictive accuracy than the full set, but still had twice the 5-y predictive accuracy than when using cytosine-adenine-guanine and age alone. Additional analysis suggested interactions and nonlinear effects that were characterized in a post hoc Cox regression model.
conclusionsMeasurement of clinical variables can substantially increase the accuracy of predicting motor diagnosis over and above cytosine-adenine-guanine and age (and their interaction). Estimated probabilities can be used to characterize progression level and aid in future studies' sample selection.
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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.