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Title: A longitudinal study of the SF-36 version 2 in Friedreich ataxia
Austin Authors: Tai, Geneieve;Corben, Louise A;Yiu, Eppie M;Delatycki, Martin B 
Affiliation: Bruce Lefroy Centre for Genetic Health Research, Murdoch Childrens Research Institute, Parkville, Victoria, Australia
School of Psychological Science, Faculty of Medicine, Nursing and Health Sciences, Monash University, Clayton, Victoria, Australia
Department of Paediatrics, University of Melbourne, Parkville, Victoria, Australia
Department of Occupational Therapy, Monash Health, Clayton, Victoria, Australia
Department of Neurology, Royal Children's Hospital, Parkville, Victoria, Australia
Department of Clinical Genetics, Austin Health, Heidelberg, Victoria, Australia
Issue Date: Jul-2017 2016-09-28
Publication information: Acta Neurologica Scandinavica 2017; 136(1): 41-46
Abstract: OBJECTIVES: The Medical Outcomes Study 36 item Short-Form Health Survey (SF-36) is one of the most commonly used patient reported outcome measure. This study aimed to examine the relationship between SF-36 version 2 (SF-36V2) summary scores and Friedreich ataxia (FRDA) clinical characteristics, and to investigate the responsiveness of the scale, in comparison with the Friedreich Ataxia Rating Scale (FARS), over 1, 2 and 3 years. MATERIALS AND METHODS: Descriptive statistics were used to examine the characteristics of the cohort at baseline and years 1, 2 and 3. Correlations between FRDA clinical characteristics and SF-36V2 summary scores were reported. Responsiveness was measured using paired t tests. RESULTS: We found significant correlations between the physical component summary (PCS) of the SF-36V2 and various FRDA clinical parameters but none for the mental component summary. No significant changes in the SF-36V2 were seen over 1 or 2 years; however, PCS scores at Year 3 were significantly lower than at baseline (-3.3, SD [7.6], P=.01). FARS scores were found to be significantly greater at Years 1, 2 and 3 when compared to baseline. CONCLUSIONS: Our findings suggest that despite physical decline, individuals with FRDA have relatively stable mental well-being. This study demonstrates that the SF-36V2 is unlikely to be a useful tool for identifying clinical change in FRDA therapeutic trials.
DOI: 10.1111/ane.12693
PubMed URL:
Type: Journal Article
Subjects: Friedreich ataxia
Health status
Rating Scales
Appears in Collections:Journal articles

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