Overview of quantitative and qualitative evidence and patient-reported outcome measures
We found only limited overlap between quantitative and qualitative studies in terms of management strategies for poor sleep quality in patients with fibromyalgia. While quantitative studies assessed a wide range of diverse pharmacological and non-pharmacological interventions, the qualitative studies focused on people’s experience of using medications and undertaking Mind–body Ex training and multidisciplinary training to improve sleep quality. We did not identify other qualitative evidence on people’s experience of other forms of exercise or non-pharmacological procedures to tackle fibromyalgia-related sleep problems. In general, qualitative studies reported that participants felt the need to take medications to address the poor quality of their sleep and to ‘continue to have a life’ but were also concerned about the occurrence of side effects. This observation is partly backed up by our synthesis of quantitative evidence, which shows that some pharmacological interventions (e.g. antioxidants, SRIs and CNS depressants) may have a positive effect on quality of life even though they do not seem to have a significant effect on sleep and are often associated with adverse effects such as dizziness, drowsiness, headache and dry mouth. Although the qualitative studies stressed the importance of taking the ‘right’ medication, the results of our quantitative synthesis did not provide reliable information on optimal pharmacological interventions for treating fibromyalgia-related sleep problems. In particular, we did not find that pharmacological interventions were superior to non-pharmacological interventions such as aerobic training in reducing sleep problems. Qualitative studies also revealed that participants who underwent Mind–body Ex training (e.g. qigong, yoga) experienced better sleep patterns (e.g. fell asleep faster) and sleep quality. These results are not consistent with the results of our NMA, which found that land-based Mind–body Ex training did not significantly improve sleep quality in people with fibromyalgia but showed some beneficial effects on their quality of life. It is also worth noting that in some qualitative and quantitative studies which focused on Mind–body Ex training, the choice of comparator treatment was questionable (e.g. waiting list). Therefore, it is challenging to know whether any positive effects experienced by some participants were a true consequence of the ‘active component’ of exercise training or the incidental result of participating in a new entertaining, structured activity. It is also worth noting that while qualitative studies reported that participants felt that the beneficial effects of exercise decreased over time, quantitative studies failed to provide information on the sustainability of treatment effects as they were often of short duration (3 months).
The findings of our qualitative synthesis also indicate that people with fibromyalgia welcome flexible and tailored advice for developing strategies to manage their sleep problems, as it is possible that certain interventions may work better for some but not for all. Our quantitative studies provided only aggregate data; therefore, analyses of relevant patient subgroups or at an individual level proved unfeasible.
Qualitative studies also reported that participants identified hormonal factors, such as menopause, and the quality of the sleep environment as important elements that could affect sleep. Our PROMs analysis also identified the characteristics of the sleep environment as a relevant domain in the measurement of sleep. Characteristics of the sleep environment were not considered in the quantitative studies. Providing choices and adapting interventions to the needs of fibromyalgia patients were also key implications of our qualitative data. Both qualitative and quantitative studies focused on adults, specifically on middle-aged women from high-income countries, making it difficult to generalise our findings to the wider fibromyalgia population.
While the characteristics of current validated PROMs for fibromyalgia vary, our analysis shows that the range of items included in each PROM captures constructs associated with sleep quality and may be considered conceptually similar. However, it is possible that outcomes which matter most to people with fibromyalgia-related sleep problems, as highlighted by our qualitative synthesis (i.e. the relationship between poor sleep and other fibromyalgia symptoms, feelings of failure and frustration and fear of going to bed), are not fully represented by the items included in the identified PROMs, which focus on items that measure primarily sleep maintenance and sleep disturbance. It is also questionable whether current PROMs measure sleep outcomes that are most relevant for children and adolescents with fibromyalgia as they have not been validated in these populations. In general, because of the lack of data on ethnicity and socio–demographic factors, it is unclear whether the current PROMs validation studies are truly representative of the wider and diverse fibromyalgia patient population.
Implications for practice and further research
The current evidence indicates that poor sleep is a common and profoundly disabling problem for people with fibromyalgia, which has negative consequences on their general health and well-being and impacts on their ability to perform activities of daily living. There is a suggestion that some forms of exercise training, psychological and behavioural therapy and some pharmacological treatments may play a role in improving fibromyalgia-related sleep problems and/or patients’ quality of life. However, the limitations of the current evidence base do not allow any reliable conclusions about optimal interventions for treating sleep problems in people with fibromyalgia. There is a clear need to improve the quality of existing evidence. Findings from our qualitative and quantitative syntheses also highlight the importance of involving people with fibromyalgia in future research.
- Future studies should be properly designed and include an adequate number of diverse patients to allow investigation of differences in patients’ responses, an appropriate control treatment, and a detailed description of the characteristics of the intervention to allow a better understanding of their true effects. Specifically:
- studies should have a clear research hypothesis and rationale
- patient representatives should be actively involved in the design of fibromyalgia research
- interventions should be compared with established therapies or adequate sham treatments to demonstrate their comparative efficacy and safety (e.g. sham treatment could include sub-effective low-intensity or generic, non-specific activity/practice); this would also permit the implementation of appropriate blinding procedures
- information on the severity of the disease, duration of illness, extent and type of sleep disturbances, and level of physical activity before and during treatment should be recorded
- patient populations should include all relevant age groups, including adolescents, and greater representation of historically (and currently) under-represented groups in fibromyalgia symptom management research, including people with different race/ethnic backgrounds, the elderly and men, to increase diversity and generalisability
- adherence and compliance with study protocols should be monitored and reported (including separate results for responders and non-responders)
- long-term follow-up assessments of primary outcomes to determine durability of effects, as well as maintenance of any behavioural changes (i.e. sustained participation in exercise activities) should be assessed
- consensus on the MCID for sleep quality should be established to facilitate future research in this field.
- Any attempt to harmonise the choice of PROMs (i.e. questionnaires and assessment scales) and a future consensus on which is the most appropriate to use in the field of fibromyalgia would be more than welcome. The current use of a variety of different assessment tools makes comparisons between studies methodologically challenging, generates inconsistency in the way fibromyalgia symptoms are identified and described, and creates confusion for patients. The development of COS for measuring sleep outcomes in adults and children with fibromyalgia would be beneficial for informing the development of new standardised PROMs for measuring sleep outcomes in children and adults with fibromyalgia. In the future, it is crucial that people with fibromyalgia are involved in the conception and content validation of any tool measuring sleep, ensuring that PROMs cover what matters most to patients.
- Considering that fibromyalgia is a complex clinical condition for which there is not an established cure and given the variety of interventions that have been studied and proposed to help people to manage their symptoms more effectively, healthcare professionals should be invited to consider more holistic approaches to the treatment of sleep problems tailored to patients’ individual needs.
- To increase transparency and research quality and reduce publication bias, a pre-registration or study protocol publication should be considered an essential requirement.
- Further unblinded studies comparing non-pharmacological interventions for the management of fibromyalgia-related sleep problems with no intervention or UC (including waiting list) are not needed. They are not designed to minimise the risks of comparing UC versus interventions that have anticipated benefits, and it is unlikely they may provide robust and reliable information on the effectiveness of these treatments.
- Experts in the field, in collaboration with patient representatives, should consider developing recommendations to help people with fibromyalgia to self-manage their sleep problems when high-level clinical evidence is lacking or not yet available.
Patient and public involvement
Public and patient involvement/engagement was undertaken throughout the project. The original research plan was developed in partnership with a woman who has considerable lived experience, having been diagnosed with fibromyalgia years ago and having suffered from sleep problems (DD). She supported the development of the funding application, and as a co-applicant was actively involved in all stages of the project. She steered this application from conception and ensured that the patient’s voice was heard throughout. In particular, she explained that poor sleep quality is a major troublesome symptom among people with fibromyalgia and stressed the importance of assessing PROMs of sleep quality in people with a diagnosis of fibromyalgia. As a member of the project management team, she attended all team meetings, provided constant support and feedback, commented on draft versions of this report, proofread and approved its final version and contributed to the writing of the Plain language summary.
She also invited a male patient representative (MP) to contribute to our project and ensure a more ‘inclusive and diverse representation’. Both patient partners (DD and MP) contributed to major project decisions including the choice of outcome measures and the categorisation of interventions. They also provided advice in terms of interpreting the findings, asking for information to be easily accessed by other patients to aid self-management, and making recommendations for dissemination activities.
They were inspiring and instrumental in shaping our dissemination plan, especially providing advice on how to disseminate our findings to patients and the public. They facilitated our communication with relevant charities and healthcare organisations in this clinical area such as Versus Arthritis and Fibromyalgia Action UK, which were actively involved in our project and are now helping disseminate our findings.
Furthermore, through their established links with relevant PPI, volunteer and social media groups we are planning to reach wider, diverse audiences. They will liaise with the Fibro Friends Support Group, the Nottingham University Hospitals NHS Trust PPI group and the Versus Arthritis PPI and volunteers’ group. Contact with these groups will allow us to collect ideas/suggestions on the best ways to disseminate our findings and make them accessible and suitable for different audiences.
Under the leadership of our patient partners and in collaboration with representatives of Versus Arthritis and Fibromyalgia UK, we are planning to host a ‘Meet the Team’ event, which will be open to patients/public and academic researchers – encouraging involvement from both sides and showcasing what ‘the project’ looks like from both perspectives, offering an opportunity to discuss the project findings and collect views and experiences. This event will be advertised through academic and patient organisation channels and social media groups (e.g. the Nottingham University Hospitals NHS Trust PPI group and the Fibro Friends Support Group). Versus Arthritis will invite their Fellows and PhD students, along with their own patient/volunteering groups. We are hoping that this will provide a ‘taster’ of our research and open channels for sharing information, as well as create opportunities for future dissemination activities and for establishing fruitful relationships beyond the end of the project. This event will ‘showcase’ the symbiotic relationship between patients and researchers, showing the importance and utmost need for this: precisely, how it makes the process of research more relevant and, therefore, more applicable to the ‘real world’ that patients have to live in. The whole team has benefitted from this experience, not only for research reasons but for ongoing friendships and possible future research applications. Below is a statement from DD as she felt it was important to add how her involvement enhanced the research and how she felt about the whole experience.
Right from the very beginning, the team were friendly and open and asked for my opinion on all aspects of the proposed research. I was included and given the opportunity to provide feedback to all discussions; being asked to speak first was very empowering and encouraged me to open up and share ‘real life’ symptoms with the team. I raised the point of non-pharmacological outcomes being included because waiting times to see a consultant are at an all-time high. This means patients are having to self-manage their conditions for longer periods. Thus, any research that highlights which interventions ‘work’ best is regarded as a priority now. I really feel that I need to champion this research team and show other patients and the public what a ‘gold standard’ experience looks like. I hope that showing this aspect in our dissemination activities will encourage more people to become involved in research.
Equality, diversity and inclusion
This is an evidence synthesis and NMA. The scope of the project was defined by the NIHR Health Technology Assessment Programme, which is committed to promoting equality, diversity and inclusion principles in research.
Fibromyalgia is more common in women and adults; however, men are often underdiagnosed. Fibromyalgia symptoms including sleep problems can impact significantly on a person’s ability to carry out normal day-to-day activities. Therefore, people with this condition may be considered under the disability position of the Equality Act (2010).
The team involved in this research project included people with a range of expertise and background.