Literature DB >> 34024983

Predicting an optimal composite outcome variable for Huntington's disease clinical trials.

Daniel K Sewell1, Journey Penney1, Melissa Jay1, Ying Zhang2, Jane S Paulsen3.   

Abstract

While there is no known cure for Huntington's disease (HD), there are early-phase clinical trials aimed at altering disease progression patterns. There is, however, no obvious single outcome for these trials to evaluate treatment efficacy. Currently used outcomes are, while reasonable, not optimal in any sense. In this paper we derive a method for constructing a composite variable via a linear combination of clinical measures. Our composite variable optimizes the signal-to-noise ratio (SNR) within the context of a longitudinal study design. We also demonstrate how to induce sparsity using a soft-approximation of an L 1 penalty on the coefficients of the composite variable. We applied our method to data from the TRACK-HD study, a longitudinal study aimed at establishing good outcome measures for HD, and found that compared to the existing composite measurement our composite variable provides a larger SNR and allows clinical trials with smaller sample sizes to achieve equivalent power.

Entities:  

Keywords:  longitudinal data; mixed effects models; optimization; signal-to-noise ratio; translational science

Year:  2020        PMID: 34024983      PMCID: PMC8132919          DOI: 10.1080/02664763.2020.1759034

Source DB:  PubMed          Journal:  J Appl Stat        ISSN: 0266-4763            Impact factor:   1.416


  4 in total

1.  Indexing disease progression at study entry with individuals at-risk for Huntington disease.

Authors:  Ying Zhang; Jeffrey D Long; James A Mills; John H Warner; Wenjing Lu; Jane S Paulsen
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2011-08-19       Impact factor: 3.568

2.  Unified Huntington's Disease Rating Scale: reliability and consistency. Huntington Study Group.

Authors: 
Journal:  Mov Disord       Date:  1996-03       Impact factor: 10.338

3.  Predictors of phenotypic progression and disease onset in premanifest and early-stage Huntington's disease in the TRACK-HD study: analysis of 36-month observational data.

Authors:  Sarah J Tabrizi; Rachael I Scahill; Gail Owen; Alexandra Durr; Blair R Leavitt; Raymund A Roos; Beth Borowsky; Bernhard Landwehrmeyer; Chris Frost; Hans Johnson; David Craufurd; Ralf Reilmann; Julie C Stout; Douglas R Langbehn
Journal:  Lancet Neurol       Date:  2013-05-09       Impact factor: 44.182

4.  Motor, cognitive, and functional declines contribute to a single progressive factor in early HD.

Authors:  Scott A Schobel; Giuseppe Palermo; Peggy Auinger; Jeffrey D Long; Shiyang Ma; Omar S Khwaja; Dylan Trundell; Merit Cudkowicz; Steven Hersch; Cristina Sampaio; E Ray Dorsey; Blair R Leavitt; Karl D Kieburtz; Jeffrey J Sevigny; Douglas R Langbehn; Sarah J Tabrizi
Journal:  Neurology       Date:  2017-11-15       Impact factor: 9.910

  4 in total

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