The Diagnostic and Statistical Manual for Mental Disorders, Fifth Edition (DSM-5), recommends the World Health Organization Disability Assessment Schedule (WHODAS) 2.0 for routine clinical use. We tested the utility of the 12-item WHODAS 2.0 in prodromal Huntington disease. Using data from 726 participants and 630 companions over a 3-year follow-up, linear mixed models were fitted to test (1) baseline and longitudinal differences by progression group; (2) participant and companion differences within each group; and (3) sensitivity of the 12-item WHODAS in comparison to the 36-item WHODAS and the Total Functional Capacity (TFC) score from the Unified Huntington's Disease Rating Scale. Participants showed baseline group differences whereas companions showed baseline and longitudinal group differences. Companions reported worse functional decline over time than participants as the disease progresses. The 12-item WHODAS detected longitudinal change better than the 36-item WHODAS and the TFC in the medium progression group. Results suggest the 12-item WHODAS 2.0 can detect baseline and longitudinal differences in prodromal HD and may be useful in HD clinical trials.
|Number of pages
|EJHG : European journal of human genetics : the official journal of the European Society of Human Genetics.
|Early online date
|4 Mar 2015
|Published - 2015
We thank the PREDICT-HD sites, the study participants, the National Research
Roster for Huntington Disease Patients and Families, the Huntington’s Disease
Society of America and the Huntington Study Group. This publication was
supported by the National Center for Advancing Translational Sciences, and the
National Institutes of Health (NIH), through Grant 2 UL1 TR000442-06.
The content is solely the responsibility of the authors and does not necessarily
represent the official views of the NIH. This research is supported by the
National Institutes of Health, National Institute of Neurological Disorders and
Stroke (5R01NS040068) awarded to Dr Paulsen, CHDI Foundation, Inc
(A3917) awarded to Dr Paulsen, Cognitive and Functional Brain Changes in
Preclinical Huntington’s Disease (HD) (5R01NS054893) awarded to Dr
Paulsen, 4D Shape Analysis for Modeling Spatiotemporal Change Trajectories
in Huntington’s (1U01NS082086), Functional Connectivity in Premanifest
Huntington’s Disease (1U01NS082083), and Basal Ganglia Shape Analysis and
Circuitry in Huntington’s Disease (1U01NS082085).