When the evidence is lacking, ways to proceed when the main result of systematic reviews are- more research needed.

Article type
Authors
Ahlberg M1, Bohm-Starke N2, Dahlberg J3, Hellberg C1, Höistad M1, Österberg M1, Pukall C4, Tranæus S1, Tranæus S5
1SBU, Stockholm, Sweden
2Department of Clinical Sciences, Division of Obstetrics and Gynecology, Karolinska Institute, Stockholm, Sweden
3 University Hospital of Umeå, Umeå, Sweden
4Department of Psychology, Queen’s University, Kingston, Ontario, Canada
5Faculty of Odontology, Malmö University, Malmö, Sweden
Abstract
Background:
For some diseases, there is limited scientific guidance regarding treatment effects or diagnostic accuracy, thus leaving clinicians and patients with limited support and guidance. Two such diseases, assessed by the Swedish Agency for Health Technology Assessment and Assessment of Social Services (SBU), are treatments and diagnostics for provoked vestibulodynia and lipedema. The main reasons that no reliable conclusions could be made were the following:
• Only a few trials, with small study samples, have been conducted.
• The studies measured different outcomes, used different instruments, and measured at different times.
Objectives: To promote more focused research on these diseases using two different methods. For provoked vestibulodynia, an international core outcome set (COS) was developed, and for lipedema, a project to prioritize research questions was initiated.
Methods:
For the COS project, the method described in the Core Outcome Measures in Effectiveness Trials (COMET) handbook was used; the project included 221 participants from 22 countries. To give guidance regarding measurement tools, the COnsensus-based Standards for the selection of health Measurement Instruments (COSMIN) guideline is used. In the lipedema project, the James Lind alliance method was used; the project included 130 participants from Sweden.
Results:
The COS project resulted in 6 outcomes that were raised as being of critical importance to patients, researchers, and clinicians, some of which were not frequently used in clinical trials, such as pain-related interference in one's life.
The prioritization project highlighted 10 research questions important to patients as well as clinicians, such as diagnostic criteria, liposuction, and hormonal treatments.
Conclusions:
When conclusive results are lacking from systematic reviews, there are ways to further assist the research community. Systematic review producers have a unique position to give guidance where prioritization of research questions could be important or where a COS needs to be developed.
Relevance and importance to patients: By including patients as equivalent stakeholders in different projects when evidence is lacking, new research can be focused to match their needs.