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dc.contributor.authorLong, JD
dc.contributor.authorPaulsen, JS
dc.date.accessioned2020-12-22T04:17:53Z
dc.date.available2020-12-22T04:17:53Z
dc.date.issued2015-10-01
dc.identifier.citationLong, J. D. & Paulsen, J. S. (2015). Multivariate prediction of motor diagnosis in Huntington's disease: 12 years of PREDICT-HD. MOVEMENT DISORDERS, 30 (12), pp.1664-1672. https://doi.org/10.1002/mds.26364.
dc.identifier.issn0885-3185
dc.identifier.urihttp://hdl.handle.net/11343/258037
dc.description.abstractBACKGROUND: It 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. METHODS: One 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. RESULTS: Adding 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. CONCLUSIONS: Measurement 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.
dc.languageEnglish
dc.publisherWILEY
dc.rights.urihttps://creativecommons.org/licenses/by/4.0
dc.titleMultivariate prediction of motor diagnosis in Huntington's disease: 12 years of PREDICT-HD
dc.typeJournal Article
dc.identifier.doi10.1002/mds.26364
melbourne.affiliation.departmentPsychiatry
melbourne.affiliation.departmentObstetrics and Gynaecology
melbourne.source.titleMovement Disorders
melbourne.source.volume30
melbourne.source.issue12
melbourne.source.pages1664-1672
dc.rights.licenseCC BY
melbourne.elementsid1022161
melbourne.contributor.authorKomiti, Angela
melbourne.contributor.authorGoh, Anita
melbourne.contributor.authorLoi, Samantha
dc.identifier.eissn1531-8257
melbourne.accessrightsOpen Access


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