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- Title
The Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS) Questionnaire: evaluation of measurement properties.
- Authors
Wiklund, Ingela; Raluy-Callado, Mireia; Stull, Donald; Jangelind, Yvonne; Whiteman, David; Chen, Wen-Hung
- Abstract
Purpose: This study was to conduct the psychometric validation of the patient and parent versions of the Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS). Methods: Data collected in a 53-week placebo-controlled multinational trial were used to evaluate item performance and reliability, validity, and ability to detect change of the six HS-FOCUS function domains. Results: HS-FOCUS was completed by 49 patients above 12 years old and 84 parents. Floor effects and high average inter-item correlations suggested that some items were less informative or redundant. For both patients and parents, the internal consistency and test-retest reliability met the >0.70 criteria for all domains except for the breathing, sleeping, and schooling/work in patients. Construct validity showed moderate to high correlations with CHAQ, CHQ, and HUI3 in activity-related concepts. Significant differences in domain scores were found in most domains among severity in disability measured by CHAQ DIS. Significant differences in HS-FOCUS change scores were found in patients whose CHAQ DIS score also changed. Conclusions: Psychometric validation of the HS-FOCUS demonstrates it is a reliable, valid, and responsive instrument that can be applied in clinical trials or disease registries. Findings on the individual item performance suggest some items could be removed without compromising its validity.
- Subjects
LYSOSOMAL storage diseases; CLINICAL trials; PSYCHOMETRICS; RELIABILITY (Personality trait); MEDICAL informatics; MUCOPOLYSACCHARIDOSIS II; HEALTH outcome assessment
- Publication
Quality of Life Research, 2013, Vol 22, Issue 4, p875
- ISSN
0962-9343
- Publication type
Article
- DOI
10.1007/s11136-012-0196-5