Journal article
WHODAS 2.0 in prodromal Huntington disease: measures of functioning in neuropsychiatric disease
European journal of human genetics : EJHG, Vol.22(8), pp.958-963
08/2014
DOI: 10.1038/ejhg.2013.275
PMCID: PMC4350592
PMID: 24327189
Abstract
Clinical trials to improve day-to-day function in Huntington disease (HD) require accurate outcome measures. The DSM-5 recommends the World Health Organization Disability Assessment Schedule (WHODAS) 2.0 for use in neuropsychiatric disorders. The DSM-5 also states proxy measures may be useful when cognitive function may be impaired. We tested WHODAS participant and companion ratings for differences in baseline and longitudinal function in three prodromal HD groups and a control group. Participants with prodromal HD were stratified by disease progression (low, medium, and high disease burden) based on their cytosine-adenine-guanine (CAG)-age product (CAP) score. Participant (N=726) and companion (N=630) WHODAS scores were examined for group differences, and for participant versus companion differences using linear mixed effects regression and Akaike's information criterion to test model fit. We also compared WHODAS with the Total Functional Capacity (TFC) scale. At baseline, functioning on the WHODAS was rated worse by participants in the high group and companions compared with controls. For longitudinal changes, companions reported functional decline over time in the medium and high groups. In simultaneous analysis, participant and companion longitudinal trajectories showed divergence in the high group, suggesting reduced validity of self-report. The WHODAS showed greater longitudinal difference than the TFC in the medium group relative to controls, whereas the TFC showed greater longitudinal difference than WHODAS in the high group. Results suggest the WHODAS can identify baseline and longitudinal differences in prodromal HD and may be useful in HD clinical trials. Companions may provide more accurate data as the disease progresses.
Details
- Title: Subtitle
- WHODAS 2.0 in prodromal Huntington disease: measures of functioning in neuropsychiatric disease
- Creators
- Nancy R Downing - College of Nursing, University of Iowa, Iowa City, IA, USAJi-In Kim - Department of Psychiatry, Roy J. and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, IA, USAJanet K Williams - College of Nursing, University of Iowa, Iowa City, IA, USAJeffrey D Long - 1] Department of Psychiatry, Roy J. and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, IA, USA Department of Biostatistics, College of Public Health, University of Iowa, Iowa City, IA, USAJames A Mills - Department of Psychiatry, Roy J. and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, IA, USAJane S Paulsen - 1] Department of Psychiatry, Roy J. and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, IA, USA Department of Neurology, Roy J. and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, IA, USA Department of Psychology, University of Iowa, Iowa City, IA, USA
- Resource Type
- Journal article
- Publication Details
- European journal of human genetics : EJHG, Vol.22(8), pp.958-963
- DOI
- 10.1038/ejhg.2013.275
- PMID
- 24327189
- PMCID
- PMC4350592
- NLM abbreviation
- Eur J Hum Genet
- ISSN
- 1018-4813
- eISSN
- 1476-5438
- Publisher
- England
- Grant note
- 1U01NS082085 / NINDS NIH HHS R01 NS040068 / NINDS NIH HHS U01 NS082085 / NINDS NIH HHS R01 NS054893 / NINDS NIH HHS 1U01NS082086 / NINDS NIH HHS 2 UL1 TR000442-06 / NCATS NIH HHS 5R01NS054893 / NINDS NIH HHS UL1 TR000442 / NCATS NIH HHS 1U01NS082083 / NINDS NIH HHS MR/L010305/1 / Medical Research Council MR/K013041/1 / Medical Research Council MR/L501517/1 / Medical Research Council NS040068 / NINDS NIH HHS U01 NS082083 / NINDS NIH HHS U01 NS082086 / NINDS NIH HHS G0902227 / Medical Research Council
- Language
- English
- Date published
- 08/2014
- Academic Unit
- Psychiatry; Psychological and Brain Sciences; Biostatistics; Nursing
- Record Identifier
- 9984064134402771
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