Dutch juvenile idiopathic arthritis patients, carers and clinicians create a research agenda together following the James Lind Alliance method: a study protocol

Schoemaker, Casper; Armbrust, Wineke; Swart, Joost F.; Vastert, Sebastiaan J.; Loosdregt, Jorg van; Verwoerd, Anouk; Whiting, Caroline; Cowan, Katherine; Olsder, Wendy; Versluis, Els; Vliet, Rens van; Fernhout, Marlous; Bookelman, Sanne; Cappon, Jeannette; M, van den Berg; Schatorjé, Ellen; Müller, P; Kamphuis, Sylvia; Boer, Joke de; Lelieveld, Otto T H M; Net, Janjaap van der; Jongsma, Karin; Rensen, Annemiek van; Dedding, Christine; Wulffraat, Nico

doi:10.1186/s12969-018-0276-3

Cited by 25 publications

(23 citation statements)

References 24 publications

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“…Patients and parents should have an active role in choosing personal targets as well. In this respect, the lack of participation of parent or patient representatives was a limitation of the work of the Task Force thus far [29,30]. Fortunately, the Task Force insisted that with the next iteration, parents and patients will be included [1].…”

Section: Resultsmentioning

confidence: 99%

Treating juvenile idiopathic arthritis to target: what is the optimal target definition to reach all goals?

2020

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In 2018, an international Task Force formulated recommendations for treating Juvenile Idiopathic Arthritis (JIA) to target. The Task Force has not yet resolved three issues. The first issue is the lack of a single "best" target. The Task Force decided not to recommend the use of a specific instrument to assess inactive disease or remission. Recent studies underscore the use of a broad target definition. The second issue is the basic assumption that a treatment aggressively aimed at the target will have 'domino effects' on other treatment goals as well. Thus far, this assumption was not confirmed for pain, fatigue and stiffness. The third issue is shared decision-making, and the role of individual patient targets. Nowadays, patients and parents should have a more active role in choosing targets and their personal treatment goals. In our department the electronic medical records have been restructured in such a way that the patient's personal treatment goals with a target date appears on the front page. The visualization of their specific personal goals helps us to have meaningful discussions on the individualized treatment strategy and to share decisions. In conclusion, a joint treat to target (T2T) strategy is a promising approach for JIA. The Task Force formulated valuable overarching principles and a first version of recommendations. However, implementation of T2T needs to capture more than just inactive disease. Patients and parents should have an active role in choosing personal targets as well.

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Section: Resultsmentioning

confidence: 99%

Treating juvenile idiopathic arthritis to target: what is the optimal target definition to reach all goals?

2020

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“…Several PSPs have been completed for common musculoskeletal conditions [13][14][15][16][17], however none addressed rare musculoskeletal disorders. This PSP applied the JLA method to a group of rare musculoskeletal disorders in adults -FD/MAS, XLH and OI, in order to identify the most important research directions in diagnosis, treatment and long-term management.…”

Section: Introductionmentioning

confidence: 99%

Rare musculoskeletal diseases in adults: a research priority setting partnership with the James Lind Alliance

Mickute

Staley²,

Delaney³

et al. 2020

Orphanet J Rare Dis

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Background Osteogenesis imperfecta, fibrous dysplasia/McCune-Albright syndrome and X-linked hypophosphatemia are three rare musculoskeletal diseases characterised by bone deformities, frequent fractures and pain. Little high-quality research exists on appropriate treatment and long-term management of these conditions in adults. This is further worsened by limited research funding in rare diseases and a general mismatch between the existing research priorities and those of the patients. This partnership adopted the James Lind Alliance approach to identify the top 10 research priorities for rare musculoskeletal diseases in adults through joint patient, carer and healthcare professional collaboration. Results The initial survey for question collection recruited 198 respondents, submitting a total of 988 questions. 77% of the respondents were patients with a rare musculoskeletal disease. Following out-of-scope question exclusion, repeating query grouping and scientific literature check for answers, 39 questions on treatment and long-term management remained. In the second public survey, 220 respondents, of whom 85% were patients with a rare musculoskeletal disease, their carers, relatives or friends, prioritised these uncertainties, which allowed selection of the top 25. In the last stage, patients, carers and healthcare professionals gathered for a priority setting workshop to reach a consensus on the final top 10 research priorities. These focus on the uncertainties surrounding appropriate treatment and holistic long-term disease management, highlighting several aspects indirect to abnormal bone metabolism, such as extra-skeletal symptoms, psychological care of both patients and their families and disease course through ageing. Conclusions This James Lind Alliance priority setting partnership is the first to investigate rare bone diseases. The priorities identified here were developed jointly by patients, carers and healthcare professionals. We encourage researchers, funding bodies and other stakeholders to use these priorities in guiding future research for those affected by rare musculoskeletal disorders.

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“…Some bodies of evidence can be combined in a mixed-methods research design with great ease while, for other combinations, additional efforts need to be made. For this kind of research that takes interests and values into account, stakeholder involvement is clearly a requisite [ 36 , 38 ]. We hope this approach helps researchers and policy-makers to recognise synergies and dilemmas between different bodies of evidence.…”

Section: Resultsmentioning

confidence: 99%

Four normative perspectives on public health policy-making and their preferences for bodies of evidence

Schoemaker

Loon

Achterberg

et al. 2020

Health Res Policy Sys

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Calls for evidence-informed public health policy-making often ignore that there are multiple, and often competing, bodies of potentially relevant evidence to which policy-makers have recourse in identifying policy priorities and taking decisions. In this paper, we illustrate how policy frames may favour the use of specific bodies of evidence. For the sixth Dutch Public Health Status and Foresight report (2014), possible future trends in population health and healthcare expenditure were used as a starting point for a deliberative dialogue with stakeholders to identify and formulate the most important societal challenges for the Dutch health system. Working with these stakeholders, we expanded these societal challenges into four normative perspectives on public health. These perspectives can be regarded as policy frames. In each of the perspectives, a specific body of evidence is favoured and other types of evidence are neglected. Crucial outcomes in one body may be regarded as irrelevant from other perspectives. Consequently, the results of research from a single body of evidence may not be helpful in the policymaking processes because policy-makers need to account for trade-offs between all competing interests and values. To support these policy processes, researchers need to combine qualitative and quantitative methodologies to address different outcomes from the start of their studies. We feel it is time for the research community to repoliticise the idea of evidence use and for policy-makers to demand research that helps them to account for all health-related policy goals. This is a prerequisite for real evidence-informed policy-making.

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Dutch juvenile idiopathic arthritis patients, carers and clinicians create a research agenda together following the James Lind Alliance method: a study protocol

Cited by 25 publications

References 24 publications

Treating juvenile idiopathic arthritis to target: what is the optimal target definition to reach all goals?

Treating juvenile idiopathic arthritis to target: what is the optimal target definition to reach all goals?

Rare musculoskeletal diseases in adults: a research priority setting partnership with the James Lind Alliance

Four normative perspectives on public health policy-making and their preferences for bodies of evidence

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