The Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS) Questionnaire: item reduction and further validation

WiklundI.; Raluy-CalladoM.; ChenW.-H.MuenzerJ.FangJ.WhitemanD.

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The Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS) Questionnaire: item reduction and further validation

机译：The Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS) Questionnaire: item reduction and further validation

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Purpose: The Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS) Questionnaire is a patient and parent-completed disease-specific instrument used in Hunter syndrome (mucopolysaccharidosis II), a rare paediatric progressive multi-systemic lysosomal storage disease. The objective of this study was to shorten the number of items of the Questionnaire to reduce response burden while maintaining its content validity.Methods: Data collected in a clinical trial were used. An iterative process helped identifying redundant or low performing items based on content validity and psychometric properties. Validation on the retained items was assessed using patients and parent’s responses in terms of reliability, validity and responsiveness.Results: The HS-FOCUS was completed by 49 patients and 84 parents. Items were mainly removed owing to high floor effects, high inter-item correlations (>0.80) or inadequate content. The shortened patient and parent versions (18 and 21 items) each contained five function domains. Internal consistency and test–retest reliability were >0.70 for most domains, except Breathing and School/work. Concurrent validity was demonstrated by significant correlations (>0.30) with similar concepts of previously validated measures. Significant differences were found in all domain scores across levels of disability.Conclusions: The shortened HS-FOCUS is a reliable, valid and responsive measure, where burden in answering the Questionnaire was reduced without compromising its validity.

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《Quality of life research: An international journal of quality of life aspects of treatment, care and rehabilitation》 |2014年第9期|2457-2462|共6页
作者
WiklundI.; Raluy-CalladoM.; ChenW.-H.MuenzerJ.FangJ.WhitemanD.;
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EvideraLondon, United Kingdom;

EvideraBethesda, MD, United States;

Department of Pediatrics, University of North Carolina at Chapel HillChapel Hill, NC, United StatesShireLexington, MA, United StatesShireEysins, Switzerland;

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正文语种英语
中图分类医药、卫生;
关键词
Hunter syndrome; Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS); Lysosomal storage disease; Mucopolysaccharidosis type II; Patient-reported outcomes;

The Hunter Syndrome-Functional Outcomes for Clinical Understanding Scale (HS-FOCUS) Questionnaire: item reduction and further validation

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