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BACKGROUND: The growth in response-shift methods has enabled a stronger empirical foundation to investigate response-shift phenomena in quality-of-life (QOL) research; but many of these methods utilize certain language in framing the research question(s) and interpreting results that treats response-shift effects as "bias," "noise," "nuisance," or otherwise warranting removal from the results rather than as information that matters. The present project will describe the various ways in which researchers have framed the questions for investigating response-shift issues and interpreted the findings, and will develop a nomenclature for such that highlights the important information about resilience reflected by response-shift findings. METHODS: A scoping review was done of the QOL and response-shift literature (n = 1100 articles) from 1963 to 2020. After culling only empirical response-shift articles, raters characterized how investigators framed and interpreted study research questions (n = 164 articles). RESULTS: Of 10 methods used, papers using four of them utilized terms like "bias" and aimed to remove response-shift effects to reveal "true change." Yet, the investigators' reflections on their own conclusions suggested that they do not truly believe that response shift is error to be removed. A structured nomenclature is proposed for discussing response-shift results in a range of research contexts and response-shift detection methods. CONCLUSIONS: It is time for a concerted and focused effort to change the nomenclature of those methods that demonstrated this misinterpretation. Only by framing and interpreting response shift as information, not bias, can we improve our understanding and methods to help to distill outcomes with and without response-shift effects.
Asunto(s)
Ruido , Calidad de Vida , Sesgo , Humanos , Calidad de Vida/psicologíaRESUMEN
BACKGROUND: Aspirations refer to wishes, ways of defining quality of life (QOL), and life goals. Living with chronic illness likely impacts a person's life aspirations. Duchenne Muscular Dystrophy (DMD) is an inherited disorder characterized by the inevitable and progressive loss of ambulation and independence. The present cross-sectional case-control study investigated how aspirations differed between people with DMD and a stratified comparison group of nationally representative children/adults. METHODS: A web-based survey was administered October through December 2020. Recruitment was stratified by age group: 8-12, 13-17, and > = 18, reflecting the DMD disability trajectory. Aspirations were measured using qualitative (open-ended) and quantitative (closed-ended) questions. Qualitative prompts asked participants about wishes, how they would define "QOL"; and goals; answers to the prompts were then coded by six trained raters. Quantitative questions included 29 closed-ended goal-delineation items from the QOL Appraisal Profilev2. These data were analyzed using multivariate models adjusting for propensity scores (demographic differences), and testing for the effects of role (patient or comparison), age, and role-by-age interactions. RESULTS: The study sample of DMD (n = 285) and comparison (n = 292) participants provided open-text data: 577 wishes statements, 283 QOL-definition statements, and 149 goals statements. Inter-rater reliability (kappa = 0.77) reflected good agreement between different raters' codes. Results suggested that people with DMD have aspirations that differ from their peers in several important ways. Both open-text and closed-ended data in both unadjusted and adjusted analyses generally showed that people with DMD were more focused on intrinsic aspirations such as health, healthcare, and independence than their peers. Compared to non-DMD persons, DMD individuals were much less focused on financial or housing concerns, community contributions, or spiritual growth. With age, patients' aspirations focused less on extrinsic aspirations such as careers and work and increasingly emphasized emotion-oriented goals. Patients were markedly less likely to give a direct answer to the open-ended goals question. CONCLUSION: There were important differences in aspirations between people with DMD and their peers. These findings may be helpful for developing psychosocial interventions.
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BACKGROUND: The inevitable and progressive loss of independence in Duchenne Muscular Dystrophy (DMD) patients may have an impact on their siblings' life aspirations. The present cross-sectional case-control study investigated how aspirations differed between brothers and sisters of people with DMD and a stratified comparison group of nationally representative children/adults. METHODS: A web-based survey was administered October through December 2020. Aspirations were measured using qualitative (open-ended) and quantitative (closed-ended) questions. Qualitative prompts asked participants about wishes, goals, and how they define quality of life (QOL) and were coded by six trained raters. Quantitative questions included 29 closed-ended goal-delineation items from the QOL Appraisal Profilev2. These data were analyzed using multivariate models adjusting for propensity scores (demographic differences) and testing for the effects of role (sibling or comparison), age, and role-by-age interactions. RESULTS: The study sample of DMD sibling (n = 349) and comparison (n = 619) participants provided open-ended data on 968 wishes statements, 390 QOL-definition statements, and 328 goals statements. Inter-rater reliability (kappa = 0.77) reflected good agreement between raters. Results of both open-ended and closed-ended data, and of both unadjusted and adjusted analyses suggested that DMD siblings, with age, were more focused on DMD-related, family/community, intimacy, and work concerns than their peers. They were less focused on improving mood, independence, pragmatics, or subtle fine-tuning of problem-solving in life. In contrast, the comparison group was more focused on goals related to growth, purpose, and reflection. Some group differences were amplified amongst older siblings. CONCLUSION: This is the first study to evaluate DMD siblings' aspirations in comparison to their peers. While there were many similarities between groups, the differences in aspirations between DMD siblings and their peers encompassed not just DMD, family/community, and intimacy, but also more work concerns. Directions for future quantitative and qualitative research are discussed.