Content and Construct Validity, Reliability, and Responsiveness of the Rheumatoid Arthritis Flare Questionnaire: OMERACT 2016 Workshop Report.

OBJECTIVE: The Outcome Measures in Rheumatology (OMERACT) Rheumatoid Arthritis (RA) Flare Group was established to develop a reliable way to identify and measure RA flares in randomized controlled trials (RCT). Here, we summarized the development and field testing of the RA Flare Questionnaire (RA-FQ), and the voting results at OMERACT 2016. METHODS: Classic and modern psychometric methods were used to assess reliability, validity, sensitivity, factor structure, scoring, and thresholds. Interviews with patients and clinicians also assessed content validity, utility, and meaningfulness of RA-FQ scores. RESULTS: People with RA in observational trials in Canada (n = 896) and France (n = 138), and an RCT in the Netherlands (n = 178) completed 5 items (11-point numerical rating scale) representing RA Flare core domains. There was moderate to high evidence of reliability, content and construct validity, and responsiveness. Factor analysis supported unidimensionality. Rasch analysis showed acceptable fit to the Rasch model, with items and people covering a broad measurement continuum and evidence of appropriate targeting of items to people, ordered thresholds, minimal differential item functioning by language, sex, or age. A summative score across items is defensible, yielding an interval score (0-50) where higher scores reflect worsening flare. The RA-FQ received endorsement from 88% of attendees that it passed the OMERACT Filter 2.0 "Eyeball Test" for instrument selection. CONCLUSION: The RA-FQ has been developed to identify and measure RA flares. Its review through OMERACT Filter 2.0 shows evidence of reliability, content and construct validity, and responsiveness. These properties merit its further validation as an outcome for clinical trials.

Successful Stepwise Development of Patient Research Partnership: 14 Years' Experience of Actions and Consequences in Outcome Measures in Rheumatology (OMERACT).

There is increasing interest in making patient participation an integral component of medical research. However, practical guidance on optimizing this engagement in healthcare is scarce. Since 2002, patient involvement has been one of the key features of the Outcome Measures in Rheumatology (OMERACT) international consensus effort. Based on a review of cumulative data from qualitative studies and internal surveys among OMERACT participants, we explored the potential benefits and challenges of involving patient research partners in conferences and working group activities. We supplemented our review with personal experiences and reflections regarding patient participation in the OMERACT process. We found that between 2002 and 2016, 67 patients have attended OMERACT conferences, of whom 28 had sustained involvement; many other patients contributed to OMERACT working groups. Their participation provided face validity to the OMERACT process and expanded the research agenda. Essential facilitators have been the financial commitment to guarantee sustainable involvement of patients at these conferences, procedures for recruitment, selection and support, and dedicated time allocated in the program for patient issues. Current challenges include the representativeness of the patient panel, risk of pseudo-professionalization, and disparity in patients' and researchers' perception of involvement. In conclusion, OMERACT has embedded long-term patient involvement in the consensus-building process on the measurement of core health outcomes. This integrative process continues to evolve iteratively. We believe that the practical points raised here can improve participatory research implementation.

Feasibility and Domain Validation of Rheumatoid Arthritis (RA) Flare Core Domain Set: Report of the OMERACT 2014 RA Flare Group Plenary.

OBJECTIVE: The Outcome Measures in Rheumatology (OMERACT) Rheumatoid Arthritis (RA) Flare Group was established to develop an approach to identify and measure RA flares. An overview of our OMERACT 2014 plenary is provided. METHODS: Feasibility and validity of flare domains endorsed at OMERACT 11 (2012) were described based on initial data from 3 international studies collected using a common set of questions specific to RA flare. Mean flare frequency, severity, and duration data were presented, and domain scores were compared by flare status to examine known-groups validity. Breakout groups provided input for stiffness, self-management, contextual factors, and measurement considerations. RESULTS: Flare data from 501 patients in an observational study indicated 39% were in flare, with mean (SD) severity of 6.0 (2.6) and 55% lasting > 14 days. Pain, physical function, fatigue, participation, and stiffness scores averaged ≥ 2 times higher (2 of 11 points) in flaring individuals. Correlations between flare domains and corresponding legacy instruments were obtained: r = 0.46 to 0.93. A combined definition (patient report of flare and 28-joint Disease Activity Score increase) was evaluated in 2 other trials, with similar results. Breakout groups debated specific measurement issues. CONCLUSION: These data contribute initial evidence of feasibility and content validation of the OMERACT RA Flare Core Domain Set. Our research agenda for OMERACT 2016 includes establishing duration/intensity criteria and developing criteria to identify RA flares using existing disease activity measures. Ongoing work will also address discordance between patient and physician ratings, facilitate application of flare criteria to clinical care, elucidate the role of self-management, and finalize recommendations for RA flare measurement.

Updating the OMERACT filter: implications for patient-reported outcomes.

OBJECTIVE: At a previous Outcome Measures in Rheumatology (OMERACT) meeting, participants reflected on the underlying methods of patient-reported outcome (PRO) instrument development. The participants requested proposals for more explicit instrument development protocols that would contribute to an enhanced version of the "Truth" statement in the OMERACT Filter, a widely used guide for outcome validation. In the present OMERACT session, we explored to what extent these new Filter 2.0 proposals were practicable, feasible, and already being applied. METHODS: Following overview presentations, discussion groups critically reviewed the extent to which case studies of current OMERACT Working Groups complied with or negated the proposed PRO development framework, whether these observations had a more general application, and what issues remained to be resolved. RESULTS: Several aspects of PRO development were recognized as particularly important, and the need to directly involve patients at every stage of an iterative PRO development program was endorsed. This included recognition that patients contribute as partners in the research and not merely as subjects. Correct communication of concepts with the words used in questionnaires was central to their performance as measuring instruments, and ensuring this understanding crossed cultural and linguistic boundaries was important in international studies or comparisons. CONCLUSION: Participants recognized, endorsed, and were generally already putting into practice the principles of PRO development presented in the plenary session. Further work is needed on some existing instruments and on establishing widespread good practice for working in close collaboration with patients.

Patient perspective workshop: moving towards OMERACT guidelines for choosing or developing instruments to measure patient-reported outcomes.

The workshop Choosing or Developing Instruments held at the Outcome Measures in Rheumatology (OMERACT) 10 meeting was designed to help participants think about the underlying methods of instrument development. Conference pre-reading material and 3 brief introductory presentations elaborated the issues, and participants broke into discussion groups before reconvening to share insights, engage in a more general discussion of the issues, and vote on recommendations. Tradeoffs between using current imperfect measures and the long and complex process of developing new instruments were considered, together with the need for rigor in patient-reported outcome (PRO) instrument development. The main considerations for PRO instrument development were listed and a research agenda for action produced. As part of the agenda for action, it is recommended that researchers and patient partners work together to tackle these issues, and that OMERACT bring forward proposals for acceptable instrument development protocols that would meet an enhanced "Truth" statement in the OMERACT Filter.

American College of Rheumatology/European League Against Rheumatism provisional definition of remission in rheumatoid arthritis for clinical trials.

OBJECTIVE: Remission in rheumatoid arthritis (RA) is an increasingly attainable goal, but there is no widely used definition of remission that is stringent but achievable and could be applied uniformly as an outcome measure in clinical trials. This work was undertaken to develop such a definition. METHODS: A committee consisting of members of the American College of Rheumatology, the European League Against Rheumatism, and the Outcome Measures in Rheumatology Initiative met to guide the process and review prespecified analyses from RA clinical trials. The committee requested a stringent definition (little, if any, active disease) and decided to use core set measures including, as a minimum, joint counts and levels of an acute-phase reactant to define remission. Members were surveyed to select the level of each core set measure that would be consistent with remission. Candidate definitions of remission were tested, including those that constituted a number of individual measures of remission (Boolean approach) as well as definitions using disease activity indexes. To select a definition of remission, trial data were analyzed to examine the added contribution of patient-reported outcomes and the ability of candidate measures to predict later good radiographic and functional outcomes. RESULTS: Survey results for the definition of remission suggested indexes at published thresholds and a count of core set measures, with each measure scored as 1 or less (e.g., tender and swollen joint counts, C-reactive protein [CRP] level, and global assessments on a 0-10 scale). Analyses suggested the need to include a patient-reported measure. Examination of 2-year followup data suggested that many candidate definitions performed comparably in terms of predicting later good radiographic and functional outcomes, although 28-joint Disease Activity Score-based measures of remission did not predict good radiographic outcomes as well as the other candidate definitions did. Given these and other considerations, we propose that a patient's RA can be defined as being in remission based on one of two definitions: (a) when scores on the tender joint count, swollen joint count, CRP (in mg/dl), and patient global assessment (0-10 scale) are all ≤ 1, or (b) when the score on the Simplified Disease Activity Index is ≤ 3.3. CONCLUSION: We propose two new definitions of remission, both of which can be uniformly applied and widely used in RA clinical trials. We recommend that one of these be selected as an outcome measure in each trial and that the results on both be reported for each trial.

American College of Rheumatology/European League against Rheumatism provisional definition of remission in rheumatoid arthritis for clinical trials.

OBJECTIVE: Remission in rheumatoid arthritis (RA) is an increasingly attainable goal, but there is no widely used definition of remission that is stringent but achievable and could be applied uniformly as an outcome measure in clinical trials. This work was undertaken to develop such a definition. METHODS: A committee consisting of members of the American College of Rheumatology, the European League Against Rheumatism, and the Outcome Measures in Rheumatology Initiative met to guide the process and review prespecified analyses from RA clinical trials. The committee requested a stringent definition (little, if any, active disease) and decided to use core set measures including, as a minimum, joint counts and levels of an acute-phase reactant to define remission. Members were surveyed to select the level of each core set measure that would be consistent with remission. Candidate definitions of remission were tested, including those that constituted a number of individual measures of remission (Boolean approach) as well as definitions using disease activity indexes. To select a definition of remission, trial data were analysed to examine the added contribution of patient-reported outcomes and the ability of candidate measures to predict later good radiographic and functional outcomes. RESULTS: Survey results for the definition of remission suggested indexes at published thresholds and a count of core set measures, with each measure scored as 1 or less (eg, tender and swollen joint counts, C reactive protein (CRP) level, and global assessments on a 0-10 scale). Analyses suggested the need to include a patient-reported measure. Examination of 2-year follow-up data suggested that many candidate definitions performed comparably in terms of predicting later good radiographic and functional outcomes, although 28-joint Disease Activity Score-based measures of remission did not predict good radiographic outcomes as well as the other candidate definitions did. Given these and other considerations, we propose that a patient's RA can be defined as being in remission based on one of two definitions: (1) when scores on the tender joint count, swollen joint count, CRP (in mg/dl), and patient global assessment (0-10 scale) are all ≤1, or (2) when the score on the Simplified Disease Activity Index is ≤3.3. CONCLUSION: We propose two new definitions of remission, both of which can be uniformly applied and widely used in RA clinical trials. The authors recommend that one of these be selected as an outcome measure in each trial and that the results on both be reported for each trial.

Understanding social role participation: what matters to people with arthritis?

OBJECTIVE: To assess the importance of different social roles in the lives of people with osteoarthritis (OA), and satisfaction with time spent in roles and role performance, as well as the relationship of demographic, health, and psychological factors to role perceptions. METHODS: Sixty women and 27 men (age 42-86 yrs) with hip or knee OA were recruited from rehabilitation programs and community advertising. Participants completed interview-administered questionnaires measuring demographics, OA symptoms, activity limitations, and well-being (e.g., depression). They also completed the Social Role Participation Questionnaire (SRPQ) assessing the influence of arthritis on role salience and satisfaction across diverse role domains (e.g., close relationships, employment). RESULTS: Participants reported many salient roles, but low to moderate satisfaction with them related to OA. SRPQ dimensions of salience and satisfaction were distinct; satisfaction with time spent in roles and with role performance was highly correlated (r = 0.83). Lower role salience was associated with being older, having less education and income, and greater illness intrusiveness. Less satisfaction with time spent in roles due to OA was associated with being younger, greater pain, and greater illness intrusiveness, whereas less satisfaction with role performance was associated with greater illness intrusiveness and depression. CONCLUSION: This study addresses a gap -- the influence of OA on social role participation. It underscores the importance of taking into account individual perceptions of roles, and that these perceptions are multifaceted. Understanding diverse factors related to social roles may help identify individuals at risk for role difficulties and provide targets for interventions to improve role participation.

Experiences of mothers living with inflammatory arthritis.

OBJECTIVE: To describe the impact of chronic, inflammatory arthritis on parenting and to develop a conceptual framework for subsequent study of mothering. METHODS: A qualitative, grounded theory design guided data collection and analysis. In-depth interviews were conducted with a purposive sample of 12 women with either rheumatoid arthritis, ankylosing spondylitis, juvenile idiopathic arthritis, or systemic lupus erythematosus who were mothers of at least 1 child living at home. Transcripts were analyzed using a systematic approach of coding and forming concepts and key categories to construct an explanatory framework. Peer checking and member checking enhanced analytical rigor. RESULTS: Analysis of participants' experiences resulted in 4 interrelated categories describing the impact of arthritis on their role as mothers: participation in mothering tasks, best described as "sometimes I can, sometimes I can't"; different types and levels of support from others; the influence of the mother's arthritis on the family; and the challenge of balancing energy and fatigue. Individuals' arthritis story, life stage, their children's developmental stage, and daily routine described the context in which mothers experienced elements of each of the 4 main categories. CONCLUSION: Inflammatory arthritis has a dramatic impact on the experience of motherhood, with both positive and negative influences. The perspectives shared by study participants may inform practice regarding problem identification and adaptive strategies, and the explanatory model generated from the data proposes hypotheses for further study.