Reliability and Validity of the HD-PRO-Triad^TM, a Health-Related Quality of Life Measure Designed to Assess the Symptom Triad of Huntington’s Disease

Article type: Research Article

Authors: Boileau, Nicholas R.^a | Stout, Julie C.^b | Paulsen, Janes S.^{c; d; e} | Cella, David^{f; g; h} | McCormack, Michael K.^{i; j} | Nance, Martha A.^{k; l} | Frank, Samuel^m | Lai, Jin-Shei^f | Carlozzi, Noelle E.^{a; *}

Affiliations: [a] Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA | [b] School of Psychological Sciences, Monash University, Clayton, Victoria, Australia | [c] Department of Psychiatry, Carver College of Medicine, The University of Iowa, Iowa City, IA, USA | [d] Department of Neurology, Carver College of Medicine, The University of Iowa, Iowa City, IA, USA | [e] Department of Psychological and Brain Sciences, The University of Iowa, Iowa City, IA, USA | [f] Department of Medical Social Sciences, Northwestern University, Chicago, IL, USA | [g] Department of Preventive Medicine, Northwestern University, Chicago, IL, USA | [h] Northwestern University, Evanston, IL, USA | [i] Department of Pathology, Rowan University – SOM, Stratford, NJ, USA | [j] Department of Psychiatry, Rutgers University, RWJMS, Piscataway, NJ, USA | [k] Struthers Parkinson’s Center, Golden Valley, MN, USA | [l] Hennepin County Medical Center, Minneapolis, MN, USA | [m] Beth Israel Deaconess Medical Center, Boston, MA, USA

Correspondence: [*] Correspondence to: Noelle E. Carlozzi, PhD, Department of Physical Medicine and Rehabilitation, University of Michigan, North Campus Research Complex, 2800 Plymouth Road, Building NCRC B14, Room G216, Ann Arbor, MI 48109-2800, USA. Tel.: +1 734 763 8917; Fax: +1 734 763 7186; E-mail: carlozzi@med.umich.edu.

Abstract: Background:Huntington’s disease (HD), is a neurodegenerative disorder that is associated with cognitive, behavioral, and motor impairments that diminish health related quality of life (HRQOL). The HD-PRO-TRIADTM is a quality of life measure that assesses health concerns specific to individuals with HD. Preliminary psychometric characterization was limited to a convenience sample of HD participants who completed measures at home so clinician-ratings were unavailable. Objectives:The current study evaluates the reliability and validity of the HD-PRO-TRIADTM in a well-characterized sample of individuals with HD. Methods:Four-hundred and eighty-two individuals with HD (n = 192 prodromal, n = 193 early, and n = 97 late) completed the HD-PRO-TRIADTM questionnaire. Clinician-rated assessments from the Unified Huntington Disease Rating Scales, the short Problem Behaviors Assessment, and three generic measures of HRQOL (WHODAS 2.0, RAND-12, and EQ-5D) were also examined. Results:Internal reliability for all domains and the total HD-PRO-TRIADTM was excellent (all Cronbach’s α >0.93). Convergent and discriminant validity were supported by significant associations between the HD-PRO-TRIADTM domains, and other patient reported outcome measures as well as clinician-rated measures. Known groups validity was supported as the HD-PRO-TRIADTM differentiated between stages of the disease. Floor and ceiling effects were generally within acceptable limits. There were small effect sizes for 12-month change over time and moderate effect sizes for 24-month change over time. Conclusions:Findings support excellent internal reliability, convergent and discriminant validity, known groups validity, and responsiveness to change over time. The current study supports the clinical efficacy of the HD-PRO-TRIADTM. Future research is needed to assess the test-retest reliability of this measure.

Keywords: HD-PRO-TRIAD^TM, health-related quality of life, Huntington’s disease, patient reported outcome (PRO), psychometric, reliability, validity

DOI: 10.3233/JHD-170238

Journal: Journal of Huntington's Disease, vol. 6, no. 3, pp. 201-215, 2017