{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T23:24:47Z","timestamp":1784676287567,"version":"3.55.0"},"reference-count":30,"publisher":"Wiley","issue":"12","license":[{"start":{"date-parts":[[2015,9,4]],"date-time":"2015-09-04T00:00:00Z","timestamp":1441324800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"crossref","award":["5R01NS040068"],"award-info":[{"award-number":["5R01NS040068"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100005725","name":"CHDI Foundation, Inc","doi-asserted-by":"crossref","award":["A6266"],"award-info":[{"award-number":["A6266"]}],"id":[{"id":"10.13039\/100005725","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100005725","name":"CHDI Foundation, Inc","doi-asserted-by":"crossref","award":["A2015"],"award-info":[{"award-number":["A2015"]}],"id":[{"id":"10.13039\/100005725","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Cognitive and Functional Brain Changes in Preclinical Huntington's Disease","award":["5R01NS054893"],"award-info":[{"award-number":["5R01NS054893"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Movement Disorders"],"published-print":{"date-parts":[[2015,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>It is well known in Huntington's disease that cytosine\u2010adenine\u2010guanine 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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>One thousand seventy\u2010eight Huntington's disease gene\u2013expanded carriers (64% female) from the Neurobiological Predictors of Huntington's Disease study were followed up for up to 12 y (mean\u2009=\u20095, standard deviation\u2009=\u20093.3) 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\u2010censored data.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Adding 34 variables along with cytosine\u2010adenine\u2010guanine and age substantially increased predictive accuracy relative to cytosine\u2010adenine\u2010guanine 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\u2010y predictive accuracy than when using cytosine\u2010adenine\u2010guanine and age alone. Additional analysis suggested interactions and nonlinear effects that were characterized in a post hoc Cox regression model.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Measurement of clinical variables can substantially increase the accuracy of predicting motor diagnosis over and above cytosine\u2010adenine\u2010guanine and age (and their interaction). Estimated probabilities can be used to characterize progression level and aid in future studies' sample selection. \u00a9 2015 The Authors. <jats:italic>Movement<\/jats:italic> Disorders published by Wiley Periodicals, Inc. on behalf of International Parkinson and Movement Disorder Society<\/jats:p><\/jats:sec>","DOI":"10.1002\/mds.26364","type":"journal-article","created":{"date-parts":[[2015,9,4]],"date-time":"2015-09-04T09:34:30Z","timestamp":1441359270000},"page":"1664-1672","source":"Crossref","is-referenced-by-count":43,"title":["Multivariate prediction of motor diagnosis in Huntington's disease: 12 years of PREDICT\u2010HD"],"prefix":"10.1002","volume":"30","author":[{"given":"Jeffrey D.","family":"Long","sequence":"first","affiliation":[{"name":"Department of Psychiatry Carver College of Medicine, The University of Iowa Iowa City IA USA"},{"name":"Department of Biostatistics College of Public Health, The University of Iowa Iowa City IA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jane S.","family":"Paulsen","sequence":"additional","affiliation":[{"name":"Department of Psychiatry Carver College of Medicine, The University of Iowa Iowa City IA USA"},{"name":"Department of Neurology Carver College of Medicine, The University of Iowa Iowa City IA USA"},{"name":"Department of Psychology The University of Iowa Iowa City IA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"PREDICT\u2010HD Investigators and Coordinators of the Huntington Study Group","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2015,9,4]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(93)90585-E"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11910-011-0215-x"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.25997"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrneurol.2014.24"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.25657"},{"key":"e_1_2_9_7_1","first-page":"452","article-title":"Rate of functional decline in Huntington's disease","volume":"54","author":"Marder K","year":"2000","journal-title":"Huntington Study Group. 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