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. © Springer Science+Business Media B.V. 2012.
CITATION STYLE
Wiklund, I., Raluy-Callado, M., Stull, D. E., Jangelind, Y., Whiteman, D. A. H., & Chen, W. H. (2013). The Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS) Questionnaire: Evaluation of measurement properties. Quality of Life Research, 22(4), 875–884. https://doi.org/10.1007/s11136-012-0196-5
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