Multivariate prediction of motor diagnosis in Huntington's disease: 12 years of PREDICT-HD.

Movement Disorders : Official Journal of the Movement Disorder Society
Jeffrey D LongPREDICT-HD Investigators and Coordinators of the Huntington Study Group

Abstract

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. 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. 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 ...Continue Reading

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Citations

Oct 7, 2016·Frontiers in Neurology·Jennifer Ashley CiarochiUNKNOWN PREDICT-HD Investigators and Coordinators of the Huntington Study Group
Nov 29, 2016·Movement Disorders : Official Journal of the Movement Disorder Society·Jeffrey D LongCristina Sampaio
Jun 23, 2018·Brain Sciences·Jennifer A CiarochiUNKNOWN PREDICT-HD Investigators and Coordinators of the Huntington Study Group
Jan 31, 2020·Biostatistics·Unkyung LeeTanya P Garcia
Jul 8, 2016·Annals of Clinical and Translational Neurology·Tanya SimuniUNKNOWN PPMI Investigators
Apr 14, 2018·Frontiers in Neurology·Jingyu LiuUNKNOWN PREDICT-HD Investigators and Coordinators of the Huntington Study Group
Jan 16, 2019·Movement Disorders : Official Journal of the Movement Disorder Society·Jane S PaulsenYing Zhang
Jun 7, 2017·Journal of Huntington's Disease·Kan LiUNKNOWN PREDICT-HD Investigators of the Huntington Study Group
Jul 1, 2019·Journal of Huntington's Disease·Fan LiUNKNOWN PREDICT-HD and ENROLL-HD Investigators of the Huntington Study Group
Apr 2, 2019·Journal of Huntington's Disease·Jennifer A CiarochiUNKNOWN PREDICT-HD Investigators and Coordinators of the Huntington Study Group
May 6, 2020·Clinical Parkinsonism & Related Disorders·Emilia M GattoClaudia Perandones
Aug 21, 2018·American Journal of Human Genetics·Jeffrey D LongJames F Gusella
Feb 27, 2017··Milan SonkaSatyananda Kashyap

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Software Mentioned

R
randomForestSRC
pec

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