Purpose Service user involvement in instrument development is increasingly recognised as important, but is often not done and seldom reported. This has adverse implications for the content validity ...of a measure. The aim of this paper is to identify the types of items that service users felt were important to be included or excluded from a new Recovering Quality of Life measure for people with mental health difficulties. Methods Potential items were presented to service users in face-to-face structured individual interviews and focus groups. The items were primarily taken or adapted from current measures and covered themes identified from earlier qualitative work as being important to quality of life. Content and thematic analysis was undertaken to identify the types of items which were either important or unacceptable to service users. Results We identified five key themes of the types of items that service users found acceptable or unacceptable; the items should be relevant and meaningful, unambiguous, easy to answer particularly when distressed, do not cause further upset, and be non-judgemental. Importantly, this was from the perspective of the service user. Conclusions This research has underlined the importance of service users' views on the acceptability and validity of items for use in developing a new measure. Whether or not service users favoured an item was associated with their ability or intention to respond accurately and honestly to the item which will impact on the validity and sensitivity of the measure.
Duchenne Muscular Dystrophy (DMD) is a severe, life-limiting and incurable condition. However, studies estimating quality of life and those measuring actual quality of life in people living with DMD ...vary considerably. This discrepancy indicates potential difficulties with assessing quality of life using common generic quality of life instruments in this rare and unique population. This study sought to document the range of themes relevant to quality of life for people with DMD by examining the published literature and additionally to investigate the themes that are relevant to quality of life for carers and the wider family. Eligible studies for the review were primary studies of any study design that reported outcomes or themes relevant to quality of life for either people with DMD, their families, or both. A review of studies identified from searching medical bibliographic sources between 2010 and 2016 found 45 relevant published studies. A thematic framework is proposed to categorise the themes identified into: i. physical; ii. psychological; iii. Social; iv. well-being domains. A final "other" domain was included to encompass themes identified from the literature that are not covered by commonly used quality of life instruments. The rich variety of themes identified from the review highlights that DMD has a complex quality of life profile which is not currently captured by standard quality of life tools that are commonly employed in the healthcare setting. The findings also highlight that the resulting impact on the quality of life of carers and wider family of people with DMD requires consideration.
Abstract
Duchenne muscular dystrophy (DMD) is an inherited X-linked neuromuscular disorder. A number of questionnaires are available to assess quality of life in DMD, but there are concerns about ...their validity. This systematic review aimed to appraise critically the content and structural validity of quality of life instruments for DMD. Five databases (EMBASE, MEDLINE, CINAHL, PsycINFO, and Cochrane Library) were searched, with supplementary searches in Google Scholar. We included articles with evidence on the content and/or structural validity of quality of life instruments in DMD, and/or instrument development. Evidence was evaluated against the Consensus-based Standards for the selection of health Measurement INstruments (COSMIN) criteria. Fifty five articles featured a questionnaire assessing quality of life in DMD. Forty instruments were extracted and 26 underwent assessment. Forty-one articles contained evidence on content or structural validity (including 37 development papers). Most instruments demonstrated low quality evidence and unsatisfactory or inconsistent validity in DMD, with the majority not featuring direct validation studies in this population. Only KIDSCREEN received an adequate rating for instrument design and a satisfactory result for content validity based on its development, yet, like the majority of PROMs, the measure has not been directly validated for use in DMD. Further research is needed on the validity of quality of life instruments in DMD, including content and structural validity studies in this population.
AbstractObjectiveThis study examines the impact of data collection method on the sociodemographic and health profile of samples of people with diabetes who complete either an online or postal ...patient-reported outcome measure (PROM) validation survey. MethodsA longitudinal survey of people with diabetes was conducted using online and postal survey versions. The survey consisted of sociodemographic and health questions, a health and self-management PROM (Health and Self-Management in Diabetes HASMID), and 5-level version of EQ-5D. Dose adjustment for normal eating Online, Diabetes UK, and social media were used to recruit online survey participants. A panel of patients at a local National Health Service Trust was randomly allocated to participate in either survey version (two-thirds to postal version). Participants were asked to complete the survey again approximately 3 months later. ResultsA total of 2784 participants completed the survey (1908 online, 876 postal). The samples (online versus postal) differed; the online sample was younger, with a larger proportion of women and respondents with type 1 diabetes. There were significant differences in sociodemographic characteristics by type of diabetes across data collection mode. The proportion of respondents who responded again at point 2 was higher in the postal sample (525 postal, 698 online). ConclusionThe sociodemographic and health profile of samples of people with diabetes differed depending on whether they completed the online or postal survey. Differences are likely due to different recruitment methods and differences in those choosing to respond to different survey versions. Future PROM validation surveys should select data collection methods carefully because these can affect sample characteristics and results.
The aim of this study was to determine the acceptability and psychometric properties of the Hypo-METRICS (Hypoglycemia MEasurement, ThResholds and ImpaCtS) application (app): a novel tool designed to ...assess the direct impact of symptomatic and asymptomatic hypoglycemia on daily functioning in people with insulin-treated diabetes.
100 adults with type 1 diabetes mellitus (T1DM, n = 64) or insulin-treated type 2 diabetes mellitus (T2DM, n = 36) completed three daily 'check-ins' (morning, afternoon and evening) via the Hypo-METRICs app across 10 weeks, to respond to 29 unique questions about their subjective daily functioning. Questions addressed sleep quality, energy level, mood, affect, cognitive functioning, fear of hypoglycemia and hyperglycemia, social functioning, and work/productivity. Completion rates, structural validity, internal consistency, and test-retest reliability were explored. App responses were correlated with validated person-reported outcome measures to investigate convergent (rs>±0.3) and divergent (rs<±0.3) validity.
Participants' mean±SD age was 54±16 years, diabetes duration was 23±13 years, and most recent HbA1c was 56.6±9.8 mmol/mol. Participants submitted mean±SD 191±16 out of 210 possible 'check-ins' (91%). Structural validity was confirmed with multi-level confirmatory factor analysis showing good model fit on the adjusted model (Comparative Fit Index >0.95, Root-Mean-Square Error of Approximation <0.06, Standardized Root-Mean-square Residual<0.08). Scales had satisfactory internal consistency (all ω≥0.5), and high test-retest reliability (rs≥0.7). Convergent and divergent validity were demonstrated for most scales.
High completion rates and satisfactory psychometric properties demonstrated that the Hypo-METRICS app is acceptable to adults with T1DM and T2DM, and a reliable and valid tool to explore the daily impact of hypoglycemia.
The Health And Self-Management In Diabetes (HASMID
) questionnaire consists of 8 attributes, 4 about quality of life, and 4 about self-management. The overall aim of this study was to rigorously ...examine the psychometric properties of the HASMID
questionnaire.
The study comprised two phases. Phase 1 identified items of the HASMID
questionnaire that potentially required rewording through consultation with a patient involvement panel and two focus groups of people with diabetes. Phase 2 involved a cross-sectional longitudinal survey where HASMID, EQ-5D-5L, health, treatment and sociodemographic questions were administered using both paper and online versions to people with diabetes. Participants were asked to complete the survey again approximately 3 months later. Psychometric analyses were undertaken to examine floor and ceiling effects, item distributions, known group differences and internal consistency. Rasch analysis was undertaken to assess differential item functioning and disordered thresholds.
Phase 1 derived five alternative wordings to items: Irritable, Affects Mealtimes, Daily Routine, Social Activities and Problem. Phase 2 achieved 2835 responses at time point 1 (n = 1944 online, n = 891 paper version) and 1243 at time point 2 (n = 533 online, n = 710 paper version). Overall the HASMID items performed well, though two alternative worded items (Irritable and Social Activities) provided additional information not fully captured by the original HASMID items.
Psychometric evaluation and Rasch analysis were used in conjunction with expert opinion to determine the final questionnaire. The application of psychometric analyses or Rasch analysis alone to inform item selection would have resulted in different items being selected for the final instrument. The benefit of a combined approach has produced an instrument which has a broader evaluation of self-management. The final validated HASMID-10 is a short self-report PRO that can be used to evaluate the impact of self-management for people living with diabetes. HASMID-10 can be scored using total summative scores, with utility and monetary values also available for use in cost-utility and cost-benefit analyses.
Duchenne muscular dystrophy is a rare, progressive, life-limiting genetic neuromuscular condition that significantly impacts the quality of life of informal caregivers. Carer quality of life is ...measured using heterogeneous self-report scales, yet their suitability for Duchenne remains unclear. This review aimed to identify and evaluate the reliability and validity of quality of life instruments in Duchenne carers.
Systematic searches were conducted in Embase, MEDLINE, CINAHL, PsycINFO, Cochrane Library and Google Scholar. Full research articles reporting data on multiple-item self-report quality of life instruments in informal Duchenne carers were included. Extracted evidence was qualitatively synthesised and evaluated, including risk of bias, against the Consensus-based Standards for the selection of health Measurement Instruments. Duchenne carer collaborators (N = 17) helped rate the instruments' content validity.
Thirty-one articles featuring thirty-two quality of life instruments were included. Content validity was rated as "inconsistent" based on very low quality evidence. For Duchenne carer collaborators, the best instrument was PedsQL Family Impact Module. Only one instrument had evidence for structural validity (rated "unsatisfactory") and measurement invariance (rated "satisfactory"). Instruments received "satisfactory" ratings for internal consistency and mixed ratings for construct validity and responsiveness. There was no evidence for reliability, measurement error, or criterion validity.
Instruments used to measure Duchenne carer quality of life have limited and often inconsistent supportive psychometric evidence. Further work must investigate instruments' measurement properties in Duchenne carers and/or the development of new tools. In the interim, we recommend considering the PedsQL Family Impact Module based on Duchenne carer ratings.
To provide a model for Public involvement (PI) in instrument development and other research based on lessons learnt in the co-production of a recently developed mental health patient reported outcome ...measure called Recovering Quality of Life (ReQoL). While service users contributed to the project as research participants, this paper focuses on the role of expert service users as research partners, hence referred to as expert service users or PI.
At every stage of the development, service users influenced the design, content and face validity of the measure, collaborating with other researchers, clinicians and stakeholders who were central to this research. Expert service users were integral to the Scientific Group which was the main decision-making body, and also provided advice through the Expert Service User Group.
During the theme and item generation phase (stage 1) expert service users affirmed the appropriateness of the seven domains of the Patient Reported Outcome Measure (activity, hope, belonging and relationships, self-perception, wellbeing, autonomy, and physical health). Expert service users added an extra 58 items to the pool of 180 items and commented on the results from the face and content validity testing (stage 2) of a refined pool of 88. In the item reduction and scale generation phase (stage 3), expert service users contributed to discussions concerning the ordering and clustering of the themes and items and finalised the measures. Expert service users were also involved in the implementation and dissemination of ReQoL (stage 4). Expert service users contributed to the interpretation of findings, provided inputs at every stage of the project and were key decision-makers. The challenges include additional work to make the technical materials accessible, extra time to the project timescales, including time to achieve consensus from different opinions, sometimes strongly held, and extra costs.
This study demonstrates a successful example of how PI can be embedded in research, namely in instrument development. The rewards of doing so cannot be emphasised enough but there are challenges, albeit surmountable ones. Researchers should anticipate and address those challenges during the planning stage of the project.
Age-related macular degeneration (AMD) is the major cause of blindness for the older population in the developed world. Geographic atrophy (GA) is an advanced form of AMD. This progressive ...degenerative disease causes loss of visual function but unlike exudative AMD there are currently no approved therapeutic treatments for GA. Instead management of the condition is through supportive care. The aim of this study was to conduct exploratory qualitative research to develop a further understanding specifically of the impact of geographic atrophy (GA) on the quality of life for both patients and their families and to explore the resources GA patients most frequently access.
Two focus groups were conducted where participants were assigned to the 'better' or 'worse' group based upon their level of visual acuity. The data were analysed using the principles of thematic analysis. Transcripts were coded using an a priori framework. Emerging themes and subthemes were added, and transcripts recoded to reflect this. Transcripts were coded by one researcher, and the emerging themes and subthemes discussed and agreed prior to transcript recoding.
Nine participants were recruited to the study (n = 5 'better group' and n = 4 'worse group'). Six overarching themes were identified. These are: experience of symptoms and understanding of GA; impact on activities; feelings and emotions; relationships and informal support; accessing formal support; and information needs.
Key findings indicated that: participants had little knowledge of the mechanisms of GA but were aware of their prognosis; activities impacted by GA included management of daily activities and reading; emotions experienced included fear and frustration, and these frequently crossed over into their relationships with others; and access to formal support was mainly practical and information needs were largely unmet, with peer support being identified as important. Management of GA should include signposting to appropriate support agencies, such as low-vision services and charities.
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