Self-management for chronic widespread pain including fibromyalgia: A systematic review and meta-analysis

Background Chronic widespread pain (CWP) including fibromyalgia has a prevalence of up to 15% and is associated with substantial morbidity. Supporting psychosocial and behavioural self-management is increasingly important for CWP, as pharmacological interventions show limited benefit. We systematically reviewed the effectiveness of interventions applying self-management principles for CWP including fibromyalgia. Methods MEDLINE, Embase, PsycINFO, The Cochrane Central Register of Controlled Trials and the WHO International Clinical Trials Registry were searched for studies reporting randomised controlled trials of interventions adhering to self-management principles for CWP including fibromyalgia. Primary outcomes included physical function and pain intensity. Where data were sufficient, meta-analysis was conducted using a random effects model. Studies were narratively reviewed where meta-analysis could not be conducted Evidence quality was rated using GRADE (Grading of Recommendations, Assessment, Development and Evaluations) (PROSPERO-CRD42018099212). Results Thirty-nine completed studies were included. Despite some variability in studies narratively reviewed, in studies meta-analysed self-management interventions improved physical function in the short-term, post-treatment to 3 months (SMD 0.42, 95% CI 0.20, 0.64) and long-term, post 6 months (SMD 0.36, 95% CI 0.20, 0.53), compared to no treatment/usual care controls. Studies reporting on pain narratively had greater variability, however, those studies meta-analysed showed self-management interventions reduced pain in the short-term (SMD -0.49, 95% CI -0.70, -0.27) and long-term (SMD -0.38, 95% CI -0.58, -0.19) compared to no treatment/usual care. There were few differences in physical function and pain when self-management interventions were compared to active interventions. The quality of the evidence was rated as low. Conclusion Reviewed studies suggest self-management interventions can be effective in improving physical function and reducing pain in the short and long-term for CWP including fibromyalgia. However, the quality of evidence was low. Future research should address quality issues whilst making greater use of theory and patient involvement to understand reported variability.


Rationale
Chronic Widespread Pain (CWP, including fibromyalgia) has a reported prevalence of up to 15% in the general population (1) causing substantial functional impairment and psychological distress. Although referrals to secondary care and specialist services are common, CWP is primarily managed in primary care (2). The treatment options available to General Practitioners (GPs) are limited. Chronic opiate prescription is common for patients with CWP, however, with growing concerns about opioid dependence (3) in addition to unique pathophysiologic characteristics of people with CWP (4) alternatives to long-term opiate use are necessary. Gabapentinoids are increasingly prescribed as alternatives to opioids, yet their utility is questionable due to a substantial side-effect profile and limited evidence for their effectiveness in CWP (5). The limitations of pharmacotherapy highlight the need for accessible non-pharmacological interventions.
The British Pain Society recently developed a care pathway for CWP (6). The pathway acknowledges the importance of supporting those with CWP to effectively self-manage, enabling them to recover and maintain quality of life, as well as reducing dependence on stretched services (6). Self-management interventions commonly have the broad aim of improving a person's health status through teaching skills to effectively manage a specific condition, often including behavioural, emotional and medical domains (7,8). Increasing the availability of effective, accessible self-management interventions may substantially improve the primary care management of CWP, and ensure secondary care services can focus on complex cases where alternative approaches may be more appropriate.
Previous systematic reviews of broad self-management in this area have focused on chronic pain in general, grouping CWP with more specific musculoskeletal conditions such as low Protocol for CWP quantitative systematic review 17/05/2018 3 back pain, knee pain, shoulder pain and osteoarthritis (7,9,10). To inform the development and use of self-management interventions accounting for CWP's unique characteristics and their impact (e.g. fatigue and psychological morbidity), a specific CWP focus is necessary.
Review evidence suggests moderate effects for single component interventions in CWP such as exercise (11), and limited effects of single psychotherapeutic approaches including guided imagery (12) and CBT (13). However, Sarzi-Puttini et al (12) suggest a multi-dimensional approach may be critical in CWP (14,15), and this is likely to apply to self-management, particularly when considering interventions for application in primary care.

Objectives
To systematically review randomised controlled trials of self-management interventions specifically targeting CWP including fibromyalgia. A secondary aim is to explore the effect of delivery modality on the effectiveness of the interventions.

Eligibility criteria
Population Adult patients receiving community, primary or secondary care for chronic widespread pain or fibromyalgia as defined by study authors (diagnostic criteria used will be extracted and documented in the review) We will include studies in patients with mixed diagnoses (e.g. osteoarthritis, fibromyalgia), provided data are reported separately for patients with our condition of interest.

Intervention
Any self-management intervention that fulfils all of the following criteria (adapted from Miles et al., (7)): i) has the broad goal of improving participants' health status or quality of life with scope for improvement in patients managing their own health.
ii) aims to increase participants' skills and knowledge and to enable participants to deploy these enhanced skills in aspects of their lives beyond the intervention iii) is directed at patients

Search strategy
We will create database specific search strategies using subject headings and text words related to CWP and fibromyalgia and self-management interventions, and database specific RCT filters. The MEDLINE strategy will be developed first, and peer reviewed by a Medical Librarian. After the MEDLINE strategy is finalised, it will be adapted to the syntax and subject headings of the other databases. There will be no language restrictions.

Data management
Titles and abstracts of all articles returned by electronic databases searches and supplemental searches will be saved in an Endnote library. Duplicate titles, where the same articles have been returned by more than one source, will be removed prior to screening.

Selection process
Screening will be performed in Covidence. Two reviewers (EM and AG) will independently screen all titles and abstracts yielded by the search against the inclusion criteria. We will obtain full papers for all titles and abstracts that appear to meet the inclusion criteria or where there is uncertainty. Two reviewers (EM and AG) will then independently assess whether these full papers meet the inclusion criteria. Disagreements will be resolved by discussion, and where applicable, arbitration by a third reviewer (MS or HE). We will record the number of papers at each stage of the process and the reasons for their exclusion.

Data collection process
We will perform data extraction into a pre-piloted Excel data extraction form. Data extracted will include: patient characteristics (e.g. age, sex, diagnostic criteria used, duration of illness, comorbidities); and elements of the Template for Intervention and Replication (TIDierR) checklist (16) including funding source. This includes physical or informational materials used in the intervention.

Risk of bias in individual studies
Risk of bias assessment will be performed by one reviewer and checked by a second. Any disagreements will be resolved by discussion, and where applicable, arbitration by a third reviewer (HE or MS). We will use the Cochrane Risk of Bias tool in accordance with guidance from the Cochrane handbook (17).

Data synthesis
We will present information in text and tables to summarise the characteristics and findings of the included studies. If studies are heterogeneous in terms of intervention and comparator, we will use a narrative approach to explore the relationship and findings both within and between the included studies.
If studies, based on clinical opinion, are sufficiently homogeneous in terms of intervention and comparator, we will conduct a meta-analysis using a random effects model. Data for cluster randomised will be treated according to the methods described in the Cochrane Handbook for Systematic Reviews of Interventions (17). For cross-over trials, we will only use the data from the first period. For dichotomous data, we will calculate risk ratios with 95% confidence intervals (CI). For continuous outcomes, we will calculate mean differences with 95% CI, or standardised mean differences with 95% if different measurement scales are used. When there are missing data, we will attempt to contact the original authors of the study to obtain the relevant missing data. Statistical heterogeneity will be tested using the Chi 2 test (significance level: 0.1) and I 2 statistic. If high levels of heterogeneity among the trials exist (I 2 ≥ 50% or P <0.1) we will explore sources of heterogeneity using subgroup analysis or sensitivity analysis.

Analysis of subgroups or subsets
If sufficient data is available: • Sensitivity analyses will be conducted to explore whether results would differ if studies at high risk of bias were excluded • Subgroup analysis will be performed for mode of delivery (e.g. individual versus group versus self-directed).
• Subgroup analysis will be performed for diagnosis of fibromyalgia versus studies of CWP more generally • Subgroup analysis will be performed for type of control condition • Subgroup analysis will be performed to explore whether results would differ depending on the components included in the interventions (e.g. interventions including psychoeducation and/or exercise)

Reporting of review
Reporting of the full systematic review will follow the Preferred reporting items for systematic reviews and meta-analyses (PRISMA) guidelines (18).