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. 2014 Apr 14:4:223.
doi: 10.7916/D8PN93NZ. eCollection 2014.

HD-PRO-TRIAD™ Validation: A Patient-reported Instrument for the Symptom Triad of Huntington's Disease

Noelle E Carlozzi  1 , David Victorson  2 , Victor Sung  3 , Jennifer L Beaumont  2 , Wendy Cheng  4 , Brian Gorin  4 , Mei Sheng Duh  4 , David Samuelson  4 , David Tulsky  5 , Sandra Gutierrez  2 , Cindy J Nowinski  2 , Allison Mueller  2 , Vivienne Shen  6 , Samuel Frank  7
Affiliations

HD-PRO-TRIAD™ Validation: A Patient-reported Instrument for the Symptom Triad of Huntington's Disease

Noelle E Carlozzi et al. Tremor Other Hyperkinet Mov (N Y). .

Abstract

Background: Few valid, disease-specific measures of health-related quality of life (HRQOL) capture the spectrum of symptoms associated with Huntington's disease (HD). The HD-PRO-TRIAD™ is a new, HD-specific, patient-reported outcome (PRO) instrument of the HD symptom triad (cognitive decline, emotional/behavioral dyscontrol, and motor dysfunction) designed for clinical research and practice. The objective was to validate the HD-PRO-TRIAD™ through a cross-sectional sample of individuals with HD and caregivers.

Methods: Development of the HD-PRO-TRIAD™ has been described elsewhere. A total of 132 individuals with HD and 40 HD caregivers, comprising 29 dyads, participated in the cross-sectional psychometric validation of this instrument. Participants provided responses to the HD-PRO-TRIAD™ and other HRQOL and disease severity instruments (EuroQOL 5D, Short Form 12, Neuro-QOL Item Banks, PROMIS Global Health, and self-reported Unified Huntington's Disease Rating Scale Total Functional Capacity and Independence Scales). Internal consistency, construct validity, and patient-caregiver proxy consistency were evaluated.

Results: Internal consistency of the three domains and overall HD-PRO-TRIAD™ instrument was supported by Cronbach's alpha values ≥0.94. Construct validity was supported by significant correlations between HD-PRO-TRIAD™ domain scores and other measures of the same domains (e.g., significant positive correlations between HD-PRO-TRIAD™ Anxiety with Neuro-QOL Anxiety), as well as slightly weaker but still strong correlations with other HRQOL instruments (e.g., HD-PRO-TRIAD™ Anxiety and UHDRS Independence; all p<0.01). Consistency between patient self-report and caregiver proxy report was supported by an intra-class correlation coefficient ≥0.92 for all three domains and the overall instrument.

Discussion: These data indicate that HD-PRO-TRIAD™ is a reliable and valid HRQOL instrument that captures the typical triad of HD symptoms.

Keywords: HD-PRO-TRIAD™; Huntington's disease; health-related quality of life; patient-reported outcome; psychometric validation; quality-of-life scale.

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Conflict of interest statement

Conflict of Interests: S.F. has received research support from the Michael J Fox Foundation and Auspex, as well as consulting fees from Merz.

Figures

Figure 1
Figure 1. Development Framework and Process for HD-PRO-TRIAD™.
1To identify the triad of symptoms relevant to an HD-specific HRQOL instrument; 2To identify HRQOL issues important to individuals with HD; 3Item development is an iterative process that includes the listed components. HD, Huntington's disease; HRQOL, Health-related quality of life.
Figure 2
Figure 2. HD-PRO-TRIAD(tm) Domains (pages 1–5)
Figure 2
Figure 2. HD-PRO-TRIAD(tm) Domains (pages 1–5)
Figure 2
Figure 2. HD-PRO-TRIAD(tm) Domains (pages 1–5)
Figure 2
Figure 2. HD-PRO-TRIAD(tm) Domains (pages 1–5)
Figure 2
Figure 2. HD-PRO-TRIAD(tm) Domains (pages 1–5)
Figure 3
Figure 3. HD-PRO-TRIAD™ Instrument and Scoring Instructions (page 6–9)
Figure 3
Figure 3. HD-PRO-TRIAD™ Instrument and Scoring Instructions (page 6–9)
Figure 3
Figure 3. HD-PRO-TRIAD™ Instrument and Scoring Instructions (page 6–9)
Figure 3
Figure 3. HD-PRO-TRIAD™ Instrument and Scoring Instructions (page 6–9)
Figure 3
Figure 3. HD-PRO-TRIAD™ Instrument and Scoring Instructions (page 6–9)
See this image and copyright information in PMC

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