Synthesis of qualitative and mixed-methods evidence evaluating the experiences and expectations of people who are treated for fibromyalgia-related sleep problems

Introduction

Value of mixed-methods qualitative studies and quantitative evidence syntheses

Historically, healthcare assessment and guideline development have been dominated by evidence syntheses that use quantitative methods with numerical data pooling.229,230 As health care has evolved towards a patient-centred model involving complex, multifactorial shared decision-making between clinicians, patients, and their families, it is important to consider the perspectives and experiences of patients in healthcare evaluations.231 Qualitative studies provide an in-depth understanding of the lived experiences of patients from a wide range of demographic and contextual backgrounds, including how patients experience symptoms related to their conditions and how their condition affects their daily lives and the lives of their family and friends, and those involved in their care. Qualitative studies can also provide an understanding of the acceptability of interventions for patients and the barriers and facilitators for intervention access and effectiveness.230

By combining quantitative and qualitative evidence syntheses in healthcare evaluations, clinical and policy decision-makers can be better informed on the management of the healthcare condition beyond issues of clinical effectiveness, to provide an understanding of the feasibility of interventions and how their implementation might vary depending on context.230

Role for qualitative studies in fibromyalgia-related sleep problems

We originally planned to systematically review existing qualitative evidence relevant to addressing our research question. However, during the application stage of our proposal, we became aware of a recently published qualitative evidence synthesis conducted by Climent-Sanz et al. in 2020, which provided an exploration of how people diagnosed with fibromyalgia experience and manage poor sleep quality.33 We therefore sought to update and extend, rather than replicate, the Climent-Sanz (2020) synthesis. The Climent-Sanz (2020) meta-synthesis findings indicated that poor sleep quality is a severe and disabling symptom associated with fibromyalgia and that prescribed treatments and self-management strategies are largely ineffective. Only studies that included adult participants and were published in English or Spanish languages were considered eligible for inclusion in the Climent-Sanz et al. synthesis. It is, therefore, unclear whether any relevant studies of the perspectives of children with fibromyalgia or studies of adults and children with fibromyalgia published in languages other than English and Spanish have been omitted from the Climent-Sanz et al. synthesis. Climent-Sanz et al. also did not report on the certainty of evidence in their synthesis. In this qualitative evidence synthesis, we sought to ascertain the experiences and expectations of people who receive treatments for fibromyalgia-related sleep problems by updating and extending the Climent-Sanz et al. synthesis to consider studies of both adults and children published in any language. We took the opportunity to identify and synthesise any newly published research into the Climent-Sanz (2020) synthesis. We also sought to apply the Grading of Recommendations Assessment, Development and Evaluation-Confidence in the Evidence from Reviews of Qualitative research tool (GRADE-CERQual) to our findings.232

Methods

Searching and identification of relevant studies

The search strategy of the Climent-Sanz et al. synthesis was developed following Peer Review of Electronic Search Strategies guideline recommendations,233 combining medical subject heading (and their equivalent in other databases) and free-text terms such as ‘fibromyalgia’, ‘sleep’, ‘sleep quality’, ‘qualitative research’.33 We repeated the search strategies from the Climent-Sanz et al. synthesis from the date of their last search (3 January 2020) to 5 November 2021. The databases searched were PubMed, Scopus, Web of Science and CINAHL. Additionally, as the current synthesis includes children, who were excluded from the Climent-Sanz et al. synthesis, the searches were re-run with text words to identify studies in children for all dates; these searches were run on 17 November 2021.

We also screened the titles and abstracts generated from our literature searches for the quantitative evidence synthesis (reported in Chapter 2) and the PROMs analysis (reported in Chapter 4) and selected any articles that seemed potentially relevant for our qualitative synthesis.

New studies identified by our updated searches were incorporated into the existing findings following current recommendations for updating qualitative evidence syntheses. The approach we have taken follows the method described by France and colleagues (2016) as ‘extending and renovating the original house’ (i.e. adding to and revising the original Climent-Sanz (2020) meta-synthesis to incorporate findings from new articles).234

We followed the eligibility criteria outlined by the Climent-Sanz (2020) synthesis; however, we extended the inclusion criteria to include studies considering children and adults published in any language. Eligible study designs included qualitative or mixed-methods research exploring the experience and/or management of sleep problems in people diagnosed with fibromyalgia.

The key eligibility criteria are summarised using the SPICE framework in Table 9.

Study selection and data extraction

The citations identified by the updated searches were independently screened between four review authors (MBe, MS, CR and MI). Full-text copies of all potentially relevant studies were retrieved and assessed for eligibility. A 10% check of all screening citations was conducted to ensure consistency between review authors. Information on the main characteristics of each identified study (e.g. aims and methods, populations involved) was extracted into a data-extraction form designed for this assessment. Data from the newly identified studies were extracted from the results sections of the included reports by a single review author (MBe or MS) and checked by a second review author for 10% of the included studies (MBe or MS).

Qualitative analysis

The analysis was conducted by two review authors (MBe and MS), who first conducted a deductive analysis by mapping the extracted data to the existing analytical themes of the ‘symptom experience’ and ‘symptom management’ components of the Symptom Management Theory (SMT)235 conceptual framework used in the Climent-Sanz (2020) synthesis.33 Any data that did not fit into the existing analysis was captured as a new theme and discussed within the team as to ‘fit’ within the SMT. The SMT provides a multidimensional framework that can be used to evaluate the patient’s experience of their symptoms. A symptom can be defined as the subjective experience reflecting changes in the biopsychosocial functioning, sensations or cognitions of the individual.236 In the SMT, symptom experience is determined by three main components: the individual’s perception of a symptom, their evaluation of the meaning of a symptom, and their response to that symptom. The relationship between these components is bidirectional; thus an individual’s evaluation of a symptom may influence their perception of the intensity of a symptom, and consequently shape their symptom response.236 We align with Climent-Sanz et al.’s position that the symptom-focused conceptual framework of the SMT is particularly useful for understanding patients’ experiences of chronic health conditions like fibromyalgia, where the main treatment goals are the alleviation and management of symptoms rather than curative treatment.

The initial reading and coding of the findings were undertaken independently by the two reviewers, and any disagreements were resolved by consensus or arbitration by our patient partners.

Quality-assessment strategy

The Critical Appraisal Skills Programme (CASP) checklist for qualitative studies was used to assess the quality of each of the newly included studies.237 To ensure consistency, two review authors (MBe and MS) independently double-assessed the risk of bias in the first 10% of the identified studies. Single assessment was performed by the same review authors for the remaining studies. Any disagreements were resolved by discussion between the two review authors. The CASP assessments for the individual studies included in the Climent-Sanz (2020) synthesis were obtained from the review authors and included in the overall assessment of study quality.

Confidence in the findings of the qualitative synthesis

To enhance the quality of the findings of the Climent-Sanz (2020)33 qualitative evidence synthesis and to integrate them with the results of our quantitative synthesis, we applied the GRADE-CERQual tool to the findings of our updated synthesis.232,238

The GRADE-CERQual approach is based on four components: the methodological limitations of included studies, the coherence of the review findings and the adequacy of data contributing to the review findings, and the relevance of the included studies to the review question. Two review authors (MI and CR) made an overall GRADE-CERQual assessment of confidence based on each thematic finding of the synthesis. Judgements on the initial assumption were that all findings were ‘high confidence’ and a reasonable representation of the phenomenon of interest, and then downgraded accordingly if there were concerns regarding any of the GRADE-CERQual components.

Findings

Description of included studies

The literature search identified a total of 61 citations. Following title and abstract screening, 29 articles were retrieved for full-text assessment. Twenty studies were excluded because they failed to meet our pre-specified inclusion criteria. Reasons for exclusion were: no suitable data reported (n = 14), ineligible study design (n = 3), ineligible study population (n = 1), full-text report not available (n = 1), and one study was available as a conference abstract only. Nine reports from eight studies were included in this synthesis. One study was identified from our literature search for our PROMs analysis239 and four studies were identified from our search for our quantitative evidence synthesis.240–243 Four reports evaluated participants’ perceptions of mind–body interventions for poor sleep quality in fibromyalgia patients.240,241,243,244 The remaining studies included one study conducted by the same lead author as the original meta-synthesis and explored the experiences and management of poor sleep quality in people with fibromyalgia.245 A further study explored the psychological functioning and psychosocial processes associated with living with fibromyalgia.239 One study focused on the experiences of fibromyalgia patients during the first COVID-19 lockdown in France (the exact dates are not reported by the study authors but correspond to weeks in April–May 2020).246 Two studies evaluated the experiences of fibromyalgia patients undergoing a 4- to 6-week non-pharmacological, multidisciplinary educational group intervention247 and a 10-week community-based group intervention involving education, exercise and sleep management.242

It is important to highlight that two of these reports by Sawynok and colleagues (2013) and Sawynok and Lynch (2014)240,241 are linked to a RCT included in our quantitative NMA, which compares the effectiveness of level 1 Chaoyi Fanhuan Qigong (CFQ) with a wait-list control for improving sleep quality in fibromyalgia patients.55 Participants from this trial were invited to participate in a further trial, described by the authors as an extension trial, in which level 2 CFQ (meditation) was added to the level 1 CFQ (movement) instruction. In the report by Sawynok and colleagues (2013), responses to a questionnaire, which invited open-ended comments on participants’ experience of the interventions, were analysed for 20 participants who took part in the extension trial. The 2014 report also analysed free-text responses to questionnaire items for the subgroup who took part in the open-label extension trial (n = 20) and all participants who completed the RCT (n = 73). It is unclear whether the 20 participants in each of the trial extension studies were the same participants. For the purposes of our analyses, we have treated these studies as comprising data from the same participant sample. Curry and colleagues (2021) also evaluated qigong as part of a mixed-methods observational study that combined quantitative measures and qualitative comments arising from open-ended comments obtained via a survey questionnaire.244 Finally, the study by Lazaridou and colleagues (2019) conducted semistructured telephone interviews with participants with fibromyalgia who had completed a non-randomised pilot study exploring the quantitative effects of yoga on sleep, pain and stress.243

A visual summary of the study screening process is provided as Figure 8. The bibliographic details of the included studies are provided in Report Supplementary Material 13.

FIGURE 8 Preferred reporting items for systematic reviews and meta-analyses flow diagram for identification of the qualitative studies.50 (Figure not reproduced here; see the original article.)

The key characteristics of the included studies are summarised in Table 10. The studies included in the Climent-Sanz (2020) synthesis were published between 2000248 and 2018.249 The studies included in our synthesis were published between 2012239 and 2021.244–246 Collectively, the studies included across both syntheses were conducted in North America (eight studies in Canada240–242,244,248,250–252 and six studies in the USA243,253–257) and Europe (five studies in England,39,247,258–260 two studies in Sweden239,261 and one study each in Finland,262 France,246 Northern Ireland249 and Spain245). One study was conducted with participants from France, Germany and the USA.263 All studies included only adult participants. In total, the studies reported data about the perspectives of 565 people with fibromyalgia. Of the included participants, 46 were men and 434 were women. The sex of the 11 participants included in the study by Teo and colleagues242 and the 73 participants who completed the RCT in the study by Sawynok 2014241 was NR. The sex of one participant included in the study by Curry and colleagues (2021)244 was not given. The youngest and oldest reported mean ages of the participants were 41 years248 and 61 years255 respectively. Only eight studies39,243,253,255,256,258,263,264 reported the participants’ race or ethnicity. The majority (80.5%) of participants in these eight studies were described as white or Caucasian. Other demographic characteristics (e.g. marital status, employment status and educational attainment level) were often NR or were reported in formats that made it difficult to make quantitative comparisons between studies.

Quality-assessment results

The majority (84.6%) of studies had a high overall CASP score, with 10243,246,247,249,252,254,259,261,263,264 scoring 10/10 for the CASP checklist items and 1239,239,245,248,250,251,253,255,256,258,260,262 scoring 9/10. The study conducted by Curry and colleagues (2021) was considered poor quality, scoring 3/10 for the CASP checklist items.244

All studies provided a clear statement of aims and considered ethical issues, and the majority (88.5%) were considered valuable and relevant to address our research question. All studies were judged to have used appropriate recruitment strategies except for the studies conducted by Curry and colleagues (2021),244 Teo and colleagues (2017)242 and Theadom and Cropley (2010).260 The recruitment strategies used in these studies were either unclear or considered only partially adequate. Three studies conducted by Curry and colleagues (2021),244 Sawynok and colleagues (2013)240 and Sawynok and Lynch (2014)241 did not consider sleep as a primary outcome and, while providing some valuable data, had less relevance for this qualitative synthesis. The authors of these three studies did not justify or discuss the choice of their study design, whereas all the remaining studies were judged to have provided adequate justification and rationale for their chosen study design. These three studies were also the only studies that were judged to have failed to have sufficiently rigorous qualitative data analysis and the authors did not provide a clear statement of their findings. It was unclear whether study authors had adequately considered the relationship between the researcher and participants in over half (53.8%) of the studies, while two studies (7.7%), conducted by Curry and colleagues (2021)244 and Arnold and colleagues (2008),253 were considered to have failed to adequately consider this relationship.

Overall findings

As stated earlier, we adopted the SMT conceptual framework used in the Climent-Sanz (2020) synthesis.33 Our findings are presented by the two pre-established global themes: the experience of poor sleep quality in fibromyalgia and management strategies for poor sleep quality in fibromyalgia. The global themes correspond to the ‘symptom experience’ and ‘symptom management strategies’ components of the SMT. The results are then presented by the four themes that were identified in the Climent-Sanz (2020) analysis and were mapped onto the SMT framework.33 We amended the name of the theme management strategies to favour sleep to management strategies to encourage sleep. We took this decision to better reflect participants’ experiences of medication-induced sleep. The presentation of findings for the global themes and themes in this report is as follows:

• The experience of poor sleep quality in fibromyalgia
  • evaluation of poor sleep quality
  • response to poor sleep quality.
• Management strategies for poor sleep quality in fibromyalgia
  • management strategies to encourage sleep
  • managing the consequences of a sleepless night.

By a process of reciprocal translation, we compared and grouped subthemes that were identified in the nine additional reports according to their shared meaning and these are presented for each relevant theme as a narrative synthesis of our findings with illustrative quotations. Most findings from the new articles were confirmatory (reciprocal) of the subthemes identified by Climent-Sanz and colleagues (2020).33 None were contradictory (refutational); however, we amended the name of one of the subthemes identified in the Climent-Sanz (2020) synthesis that is associated with the evaluation of poor sleep quality theme. We amended the subtheme name perceived effects of poor sleep quality in other symptoms of fibromyalgia to relationship between poor sleep quality and other symptoms of fibromyalgia to encompass the often-complex relationship between poor sleep quality and other fibromyalgia symptoms more fully. Furthermore, we identified a new subtheme concerning participants’ experiences of interventions to improve sleep quality, which we named experiences of interventions. Many of the included studies did not report demographic data for specific participant quotations. The contribution of each paper to the themes and subthemes is presented in Table 11.

Global symptom management theory theme 1: experience of poor sleep quality in fibromyalgia

This global theme describes the participants’ perceptions of poor sleep quality. Perceptions of good sleep quality and poor sleep quality are subjective and are likely to vary between and within individuals depending on how that person evaluates a good night’s sleep.265 For example, a good night’s sleep for one person might be continuous sleep with no or infrequent awakenings, while for another person the total number of hours spent asleep might be more important. This global SMT theme is presented as two themes: the participants’ evaluation of the meaning of their poor sleep quality and their response to poor sleep quality, which are presented in detail below with associated subthemes.

Theme 1: evaluation of poor sleep quality

In keeping with the approach taken by Climent-Sanz and colleagues (2020), and in accordance with the SMT, we conceptualised the evaluation of poor sleep quality as the meaning that a person attributes to their experience of poor sleep quality in terms of its effect, severity, temporality, cause and treatability. Three of the nine reports included in this update contributed findings to the evaluation of the poor sleep quality theme.239,245,246 Thus, a total of 16 reports contributed findings to this theme.39,239,245,246,250–252,253,256,258–264 The following subthemes were developed from analysis of participants’ descriptions.

Subtheme 1: poor sleep quality is a severe symptom of fibromyalgia. Participants frequently described poor sleep quality as a significant aspect of living with fibromyalgia, with some participants describing it as the worst symptom they experience. Being unable to sleep properly was seen as a betrayal of the body, leading to a sense of helplessness and lack of control to the extent that being able to sleep well was seen as unachievable or a ‘fantasy’.

Sleep, or lack of it, is the worst thing about this condition for me […]. It’s just another way my body has betrayed me.

I would say that it would be like a fantasy to be able to sleep well, because I sleep very badly.

I don’t feel like I can sleep. This is aging me; I can feel it.

Sleep disturbances and poor sleep maintenance were often mentioned as problems the participants experienced most often, and that the experience of being unable to stay asleep or waking frequently throughout the night would leave them feeling exhausted and frustrated. Others described problems with falling asleep or feeling unsatisfied with the quality of their sleep.

The problem is that I wake up, the problem is that when I sleep, I don’t sleep. I am sleeping, but it is as if I am awake.

Subtheme 2: relationship between poor sleep quality and other symptoms of fibromyalgia. As discussed earlier, this concept most closely resembles the concept described as ‘perceived effects of poor sleep quality in other symptoms of fibromyalgia’ described by Climent-Sanz and colleagues (2020).33 Our rationale for the coding of this concept is that our interpretation of the participants’ description of the relationship between poor sleep quality and other symptoms of fibromyalgia is complex and bidirectional, rather than a single directional impact of sleep problems on their other symptoms. We believe that this approach is in keeping with the bidirectional aspect of the SMT and the Climent-Sanz (2020) interpretation regarding poor sleep, pain and fatigue as a vicious circle in which insufficient sleep results in an increase in pain intensity the next day, which then leads to a state of fatigue that prevents sleep at night and impacts on activities of daily living. Conversely, some participants also felt that getting a good night’s sleep increased their pain due to being physically immobile while they were asleep.

It’s not just the pain and the fatigue, […] it’s the non-restorative sleep […] it’s a vicious circle because if you don’t get enough sleep, you feel pain more acutely […] you’re more tired and unable to sleep well.

It is that you wake up in the morning already with fatigue that looks like you have been run over by a truck, and you cannot handle your life.

If I sleep really well at night, I’m in much worse shape in the morning as far as pain goes … I’m not in as much pain because I’ve been moving. But then I’m a lot more tired, so I’m not going to be doing all that much.

As identified by Climent-Sanz et al. (2020), we also found that participants described the negative impacts of poor sleep on their cognitive function, including impaired ability to think and perform cognitive tasks in the workplace. In one study identified by our updated search participants described how poor sleep quality negatively impacted their mental health by causing feelings of heightened anxiety, irritability and nervousness.245 This was described as, in turn, impacting on participants’ physical health and ability to manage their symptoms. Participants also described how not being able to predict whether they would be able to sleep at night caused them to have poorer mental health.

[I fear] that I will go a little crazy […] because not sleeping gives you a lot of problems, gives you a lot of anxiety, you do not live your life with peace of mind, you have a very bad temper.

[…] there is nothing constant, it may be normal one day, and another day I can feel very tired or very nervous. The next night, I may sleep, or I may not sleep again […]. It changes every night, there is nothing constant.

As described by Climent-Sanz and colleagues (2020), we also note that participants described how their poor sleep quality impacted family members, such as causing disruption to bedfellows. Participants also described how they missed out on spending time with their family due to having to take naps to cope with fatigue brought on by their poor sleep.

I don’t hardly ever sleep with my husband any more, because I disturb his sleep so much of the time with my tossing and turning, trying to get comfortable, getting in and out of bed, because I can’t get comfortable.

Subtheme 3: beliefs about the temporality and cause of poor sleep quality. Participants described different beliefs about the causes of their poor sleep quality, ranging from the impact of other symptoms such as pain and tinnitus, to hormonal causes, such as the menopause, and working night shifts. Having to go to the bathroom was also discussed as disturbing sleep. Participants also described the relationship between sleep quality and the sleep environment, such as external noise causing poor sleep quality. Behaviours such as the consumption of stimulants like caffeine and increased physical activity levels were mentioned as additional causes of poor sleep quality.

Sometimes because of the arms and hands, because of the pain. And other times I go to the bathroom.

When you are silent is when you hear them (the tinnitus) the most. Then this […] and of course, if you wake up at two and you have this, it is very difficult to go back to sleep.

[…] everything happened more or less when my period stopped, and so I said, ‘This is menopause’. Sometimes you say, ‘This must be caused by the menopause’ […] you always look for the solution or attribute the problem to something else, yes.

Subtheme 4: meaning of good sleep quality. In keeping with the Climent-Sanz (2020) findings, we identified good sleep quality as the feeling of being rested or renewed upon waking, a feeling of disconnection or not being able to remember dreaming and having enough energy to perform daily activities. Waking with the absence of pain was also described as being related to good sleep quality.

Getting up in the morning and feeling rested, I do not care about the hours, at least open my eyes and say, ‘I have slept, I feel rested, come on, let’s start the day’.

That you do not remember what you have dreamed about, that you do not remember anything and get up and say: I feel so rested, I got up feeling so well!

That you can wake up without any pain, this would be good for me […] the best.

Theme 2: response to poor sleep quality

The second theme that was identified as relevant to the experience of poor sleep quality in fibromyalgia was the response to poor sleep quality. This is conceptualised within the SMT framework as a person’s reaction to their symptom at the physiological, psychological and social level. For example, if a person perceives (psychological level) that their symptom is serious and may cause disruption to their lives (social level), then the perceived intensity (physiological level) of that symptom may be heightened.236 In keeping with the Climent-Sanz (2020) findings, we did not identify reports of any social factors relating to the participants’ responses to poor sleep quality. Only one of the additional studies conducted by Climent-Sanz and colleagues (2021)245 contributed to the response to the poor sleep quality subtheme. Therefore, a total of six studies contributed to this subtheme.39,245,249,252,254,260 The following subthemes were identified:

Subtheme 5: feeling frustrated and like a failure. In accordance with the Climent-Sanz (2020) findings, participants described how constantly waking up is the worst thing about having fibromyalgia and how this can cause desperation and loneliness, and feelings of frustration that they are unable to sleep well. Participants also noted that they experience feelings of failure due to the negative impact that poor sleep quality has on their ability to work and perform daily activities of living. Participants also stated that they felt resentful that they must plan their lives around their sleep problems, to the extent that they sometimes ‘hate life’.

Sleep, or lack of it, is the worst thing about this condition for me.

The not sleeping and then not being able to function the next day when you need to perform at work […] when you’re being paid and you’re meant to work and you can’t function, it’s horrible, it’s really horrible because you feel like a failure.

I’m not saying my life is so exciting but when it’s, just now I think oh, I have to plan it round, oh can I have a lay in this weekend, can I do this, can I do that, I find that very hard to cope with.

Subtheme 6: Fear of going to bed. The participants expressed how the anticipation of not being able to sleep or having poor sleep, including experiencing nightmares, leads to feelings of being afraid of going to bed and being afraid of the bedroom.

It was torture for me to go to sleep because I knew that I would have nightmares, I would have aches that I could not sleep at all […]. Initially, before going to sleep, I was already worried about what was going to happen to me in the dream.

As described by Climent-Sanz and colleagues (2020), the fear of poor sleep quality has such an effect on the personal lives of some participants that they were reconsidering parenthood due to the fear that their sleep needs would be further negatively impacted to the extent that they would be unable to cope.

We’re wanting to start a family, so that’s kind of an issue regarding sleep, actually; yes, that’s a massive issue, because we’ve been wanting to start a family for ages, we’re not even doing that because I don’t think I could have that sort of sleep deprivation.

Global symptom management theory theme 2: management strategies for poor sleep quality in fibromyalgia

Within the SMT, the goal of symptom management is to avert or delay negative outcomes by identifying management strategies in response to an assessment of symptom experiences from the individual’s perspective.236 The global theme management strategies for poor sleep quality in fibromyalgia describes the strategies employed by the participants to manage their symptom experiences in response to their experience of poor sleep. The global theme is presented under the two subthemes identified by Climent-Sanz and colleagues (2020) describing participants’ management strategies to initiate and maintain sleep, and how they manage the consequences of a sleepless night.

Theme 3: management strategies to encourage sleep

We identified three new studies in the update that contributed to this theme.245–247 When added to the seven studies from the Climent-Sanz (2020) synthesis,248,252–254,258,260,262 a total of 10 studies contributed data to this theme. This theme is composed of three subthemes capturing ‘medication: from dependency to rejection’, ‘experiences of interventions’ and ‘self-management: behavioural adaptations’ as discussed below.

Subtheme 7: medication: from dependency to rejection. As with the Climent-Sanz (2020) synthesis, the participants often described using medication to assist them to fall asleep and maintain sleep. The participants described using medication indicated for insomnia and analgesia in response to sleep disturbances caused by pain to allow them to fall back asleep. Some participants felt that they benefitted from taking medication, although the importance of taking the ‘right’ medication that worked for them was highlighted as important. Medication was also discussed in terms of participants’ dependency on medication to allow them to fall asleep and alleviate their anxiety about falling asleep. The contrast between ‘normal’ sleep and medicated sleep was also discussed, with participants stating that medicated sleep feels like they ‘pass out for a while’ rather than experience sleep, although this did allow participants to feel physically rested. Other participants felt that medication was either ineffective or gradually became less effective over time and some rejected the use of medication due to concerns about side effects.

Now I am already taking Zolpidem, I do not sleep, I pass out for a while […] I am rested, because with other medicines, with other drugs that I have taken, I could not sleep.

I think that if I take it, it hurts me even more. Not in terms of sleep, but I feel worse. […]. You are full of drugs, it is not you, you are a vegetable. […] they put you to sleep, but to sleep, you’d better die now.

You get used to the drugs and after a while they no longer work.

Subtheme 8: experiences of interventions. Four of the reports identified in our update evaluated mind–body interventions to improve sleep quality. These reports included qualitative data describing the participants’ experiences of the interventions. Three reports evaluated the effects of different types of qigong.240,241,244 Qigong is a form of traditional Chinese medicine involving body–mind exercises in the form of specific slow-flowing movements, breathing techniques and meditation.266 As discussed earlier, participants in the two reports conducted by Sawynok and colleagues (2013)240 and Sawynok and Lynch (2014)241 evaluated participants’ experiences of CFQ. Curry and colleagues (2021) report the results of two observational studies (RIM1 and RIM2) of qigong practice in a sample of chronic pain patients with various underlying health conditions.244 We have analysed the comments reported by the 13 participants with fibromyalgia.

Some participants noted that they felt less pain when they practised qigong and that this had led to improved sleep quality. Improvements in sleep quality were described in terms of reduced time to sleep onset, longer sleep duration, fewer awakenings during the night, being able to fall back to sleep faster, more ‘restorative’ or ‘restful’ sleep and less need to sleep during the day. For these people, qigong allowed them to feel calmer and more relaxed. Participants reported that they had more energy and less extreme fatigue and generally felt that their overall health had improved but they noticed that symptoms quickly returned if they stopped practising qigong. Some participants felt that initial improvements lessened over time, while others did not notice any improvement in their sleep patterns or reported increased pain resulting in even greater sleep disturbance.

Qigong has given me my life back. When I started, was using a walker, couldn’t sleep and was in terrible pain. Now feel peaceful; am walking, sleeping well; pain levels have come down considerably.

[…] previously had days of extreme tiredness and slept 4 hours in afternoon, have not had to sleep during day since beginning qigong.

Felt wonderful after beginning of study, qigong was great benefit. until 3 weeks ago, woke to find fatigue and exhaustion had returned, couldn’t fall asleep.

Overall pain has increased, interfering with sleep.

Lazaridou and colleagues (2019) conducted a qualitative evaluation of group and daily individual home yoga in women with fibromyalgia.243 Fifteen women participated in semistructured telephone interviews after completing a 6-week Satyananda yoga programme. The authors describe Satyananda yoga as a holistic approach to developing an individual’s physical, mental, emotional, psychic and spiritual state of being. Participants reported experiencing reductions in their fibromyalgia symptoms, including pain and stress, and noticed positive impacts on mood, sleep and self‑confidence. Participants reported that they fell asleep faster and felt that the quality of their sleep had improved. Participants also felt that they were more alert and had more stamina during the day.

When I practiced the yoga or meditation before bed I would sleep faster.

I sleep deeper; my sleep quality is better.

In addition to the studies of mind–body interventions, Pearson and colleagues (2020) evaluated the Fibromyalgia Self-Management Programme (FSMP), which is a non-pharmacological, multidisciplinary education group intervention developed by allied health professionals within the NHS.247 The main aims of the FSMP are to provide condition-specific, patient-centred education and exercise advice and to support the development of core self-management skills through behaviour change. Patients described how they benefitted from the educational component of the intervention by understanding more about links between sleep and pain, such as the association between substance P (a modulator of pain perception) and melatonin. Participants also found the practical tips for managing their sleep through behavioural adaptations helpful, such as buying blackout curtains or changing their bed.

I mean I’ve been thinking about these things for a long time but the substance P and your melatonin and all of those sorts of things, which I was just aware of but you don’t always relate it to yourself. So that’s been very, very helpful.

Teo and colleagues (2017) conducted a feasibility study of 10-week multidisciplinary intervention involving group-based education sessions, exercise and sleep management in a small urban centre in southern British Columbia, Canada.242 Some of the participants in this study benefitted from having an organised treatment plan that involved alterations to their sleep aid therapies, such as a reduction in their medication.

He cut my medicine in half, my night time medication in half […] which enabled me to sleep better. Instead of giving me a sleep aid, he cut mine in half, which was tremendous.

Subtheme 9: self-management: behavioural adaptations. In keeping with the Climent-Sanz (2020) findings, participants described the importance of establishing regular sleep cycles or behavioural patterns that work for the individual. Strategies such as the use of earplugs, blackout curtains, buying a comfortable bed, eating at the same time of day, going to bed at the same time of night and keeping the bedroom solely as a space for sleeping were described by the participants. Some participants found it useful to try to rest during the day, while others preferred to try to physically tire themselves and avoid napping to improve sleep at night. These strategies were described as having mixed and changeable success depending on the individual person.

Well, you may have been advised to try not to eat too much or not to eat too much at night […] if you want to read, do it on the sofa, don’t go to bed […]. Everyone is different. So, each one of us must have our own tactics.

There are days when one thing works for me but then the next day I am no longer doing well. There are periods when I feel better […]. A week goes by and gets terrible again.

As with the Climent-Sanz (2020) findings, opposing strategies were employed for dealing with awakenings during the night, with some participants preferring to get out of bed while others preferred to stay in bed to try to fall back to sleep. Delaying going to bed was also discussed as a strategy to avoid waking up. Some participants stated that, while they were aware of strategies for avoiding poor sleep quality, they did not always follow the advice they were given.

I don’t go to sleep before one […] I don’t want to go at eleven or twelve because then I would already be awake at three or four.

I play with the computer and they already told me that I shouldn’t do it because this is bad for sleeping.

Theme 4: managing the consequences of a sleepless night

This theme describes how participants cope with the consequences of their poor sleep. We identified three new studies that contributed to this theme.242,245,246 When added to the seven studies from the Climent-Sanz (2020) synthesis,250,251,253–255,260,261 a total of 10 studies contributed data to this theme. The theme is composed of two subthemes that describe how participants find the balance between the benefits and side effects of medication and resting and relaxing during the day.

Subtheme 10: medication: finding the balance between benefits and side effects. This subtheme describes how participants viewed taking medication as a necessary consequence of their poor sleep, as distinct from their experiences of the effects that medication had on the quality of their sleep. The participants tended to be taking several medications for symptoms associated with fibromyalgia, including pain and depression, as well as sleep problems. For some participants, the consequences of taking medication were that they were able to manage their activities of daily living and medication allowed them to ‘continue to have a life’ but for some participants this was at the expense of experiencing side effects due to their treatment. For other participants, reducing their medication allowed them to experience better-quality sleep.

When I have been resting a while and I sleep when I’ve taken a painkiller […] and can sleep an hour and then it feels much better, then I can manage the afternoon.

I’m trying to function, so I take the various medications […] in the hopes that while it causes other problems it will at least allow me to continue to have a life.

Subtheme 11: resting and relaxing during the day. In keeping with the Climent-Sanz (2020) findings, participants described how they experienced daytime fatigue due to poor sleep and that relaxing or taking naps during the day were necessary coping strategies to deal with their fatigue. This theme is related to using rest and relaxation as a behavioural management strategy to encourage sleep, but here rest and relaxation were viewed as unavoidable consequences of having a poor night’s sleep. Some participants noted that sleeping during the day was against the advice they had received from health professionals for achieving good-quality sleep at night, but they stated that they felt they knew what worked best for themselves and preferred to develop their own coping strategies rather than follow generic guidance. Others described being unable to cope without resting during the day and that they planned their daily activities, such as providing food for family members, around their nap times. As described earlier, napping meant that participants missed out on spending time with their family, but this was described as unavoidable due to their experience of extreme fatigue. Relaxation strategies included taking hot baths with Epsom salts or lavender oil and lying down in a quiet room.

I know they advise you not to go to bed […] but I can’t physically not and I find it makes me feel better actually if I do, so for me it works better, so you I’ve learnt to do what suits me rather than what I’m told to do.

I have to lay down because if I don’t, I can’t stand it […] and sometimes I leave them food and go to lay down because my body can’t stand it […] and when I see that my husband stays there, sometimes I say ‘Wake up, I can’t’.

Assessment of confidence in the findings of the qualitative synthesis

Our confidence in the findings of this qualitative evidence synthesis is based on our GRADE-CERQual assessment. The GRADE-CERQual ratings are presented in Table 12. We rated each domain as ‘no or very minor concerns’, ‘minor concerns’, ‘moderate concerns’ or ‘serious concerns’. We rated most subthemes as moderate confidence. Findings were downgraded for ‘relevance’ because participant demographic data, including sociodemographic status, race and ethnicity, were poorly reported across studies. Findings for the experiences of interventions subtheme were also downgraded for ‘methodological limitations’ because three reports that contributed data to this subtheme were of overall poor methodological quality as assessed by the CASP checklist for qualitative studies. These included the studies conducted by Curry and colleagues (2021),244 Sawynok and colleagues (2013)240 and Sawynok and Lynch (2014).241 The methodological concerns for these studies were that the research design and data-collection methods were not appropriate, the relationship between the researcher and participants was not adequately considered, data analyses were not sufficiently rigorous, and the authors did not report clear statements of their findings.

Discussion

This update of an existing qualitative meta-synthesis integrates and summarises the findings of 9 newly identified studies with the 17 studies that were included in the original published synthesis conducted by Climent-Sanz and colleagues (2020).33 Most studies were of good methodological quality. We have moderate confidence in the findings of our qualitative synthesis except for people’s experiences of interventions, which we rated as low confidence. The resulting synthesis confirms the findings of the previous evidence synthesis that poor sleep is perceived as a severe symptom of fibromyalgia, with some participants describing poor sleep as one of the worst aspects of fibromyalgia. Participants also described how their sleep problems impacted on their family life, including being unable to share a bed with their partners and missing out on spending time with family members. Poor sleep maintenance and sleep disturbance were mentioned as the problems participants experienced most often. Our analysis also confirms that people with fibromyalgia perceive good-quality sleep in terms of having uninterrupted sleep, feeling rested or renewed upon waking, waking with an absence of fatigue and pain, and having enough energy to perform daily activities. This implies that good-quality sleep is perceived mainly in terms of sleep continuity and restorative sleep rather than the total duration of sleep; however, the meanings that people attribute to good-quality sleep are subjective and are likely to vary between individuals. As we will discuss in our PROMs analysis in Chapter 4, sleep quality is, therefore, a complex construct that is likely to require a multifactorial approach to its measurement or evaluation.

Our findings support the perception of a bidirectional relationship between pain and poor sleep described in the Climent-Sanz (2020) synthesis.33 Our findings also support the complex interconnected relationship between poor sleep, pain and fatigue. Poor sleep is associated with increased pain intensity and fatigue, with pain and fatigue further impairing sleep. The relationship between poor sleep, pain and fatigue was expressed by participants as having the biggest effect on them due to the negative impact that pain and fatigue have on people’s ability to perform activities of daily living, such as work demands and family commitments. This leads to feelings of frustration and resentment for some participants. While our data do not allow an interpretation of the direction of causality in the relationship between poor sleep and pain in fibromyalgia, there is evidence from studies of people with various chronic pain conditions and healthy controls that poor sleep is a more reliable predictor of pain than vice versa.267 It is likely that a biopsychosocial model can be applied to the understanding of the bidirectional sleep–pain relationship.268 Several central and autonomic nervous systems have been posited as the underlying physiological mechanisms involved in sleep and pain,10,268,269 while psychological factors such as pain catastrophising have been suggested as mediators in the sleep–pain relationship. In the example of pain catastrophising, it is suggested that people with chronic pain tend to think about their pain in the time leading up to attempting to go to sleep more often than people who do not have chronic pain, and that these thoughts can impair sleep onset and maintenance.270 Interestingly, several participants in our analysis described developing a fear of going to bed due to the anticipation of not being able to sleep or having poor sleep. This suggests that experiencing negative thought patterns around the time of going to sleep might be common in people with fibromyalgia and other chronic pain conditions. Similarly, several sociodemographic and socioeconomic factors, such as age, sex, education and income, as well as behavioural factors, such as physical activity, alcohol and smoking, have been associated with insufficient sleep.271 Sleep efficiency has also been identified as a predictor of pain.272 Again, it is not possible to make causal inferences, but this highlights how the relationship between sleep and pain is complex and may be mediated by several social and behavioural determinants of poor sleep.

Participants described the negative impacts that poor sleep has on their mental health, including impaired ability to perform cognitive tasks and heightened feelings of anxiety. As with the sleep–pain relationship, our data do not allow inferences about the causal direction between poor sleep and poor mental health. People with fibromyalgia are more likely to have higher levels of perceived stress and dysfunctional beliefs about sleep compared with healthy individuals.273 Higher levels of stress and dysfunctional beliefs are associated with greater sleep disturbance, daytime dysfunction, and poorer sleep quality.273 General mental health has also been identified as a risk factor for insufficient sleep.271 People with fibromyalgia may, therefore, benefit from stress management techniques and therapies that encourage the cognitive reframing of beliefs and attitudes towards sleep. Likewise, people with fibromyalgia may require workplace support to allow them to remain or re-enter the workforce due to the negative impacts that poor sleep has on cognition and fatigue.

Several beliefs about the temporality and causes of poor sleep were discussed by the participants. In addition to the beliefs described in the Climent-Sanz (2020)33 synthesis regarding working patterns and other symptoms of fibromyalgia, we were able to identify hormonal factors, such as the menopause, and the quality of the sleep environment as important causal beliefs held by the participants. There is a recognised association between perimenopause and menopause and various sleep problems.274–276 Similarly, our PROMs analysis (see Chapter 4) identified the sleep environment as a relevant domain in the measurement of sleep outcomes. It may therefore be important to consider the causal beliefs held by fibromyalgia patients as part of a holistic approach to shaping therapeutic options for poor sleep.

Our analysis confirms the Climent-Sanz (2020) findings that pharmacological treatment is often rejected by people with fibromyalgia due to side effects or a fear of developing medication dependency.33 While some participants described benefitting from medication, at least in the short term, others described how they did not feel that the quality of their sleep had improved. Instead, medication allowed participants to fall asleep and maintain sleep, but they felt that they had passed out rather than experienced good-quality sleep. Again, this highlights the complexity and subjectivity associated with the concept of ‘good-quality sleep’. That the benefits of medication appeared to lessen or disappear over time could indicate that caution should be applied to the long-term prescribing of pharmacological treatments for sleep problems in fibromyalgia patients. Our update also allowed the analysis of participants’ experiences of mind–body and multidisciplinary interventions in relation to sleep problems, which was not captured by the original meta-synthesis.33 As with pharmacological treatments, some participants felt that initial improvements lessened over time, while some experienced increased pain resulting in more sleep disturbance. Careful instruction may, therefore, be required when prescribing treatments that involve an element of physical activity to ensure avoidance of injury. That medication and other management strategies were viewed as being largely ineffective over time indicates that long-term management requires ongoing consideration.

While the participants recognised the principles of good sleep hygiene, they often described adopting maladaptive self-management behavioural adaptations and strategies to cope with poor sleep. While we agree with the authors of the Climent-Sanz (2020) synthesis that this could indicate that patients do not receive adequate information from health professionals to allow them to develop effective strategies for managing poor sleep, our updated findings reveal that individuals experience differing levels of success with various sleep-management strategies and that participants believe that they know what works best for them, even when this might contradict professional advice. This suggests that people with fibromyalgia may welcome flexible, individualised, tailored advice for developing strategies to manage poor sleep rather than a one-size-fits-all approach.

Strengths and limitations

We used established and scientifically rigorous methods for the conduct, quality assessment and reporting of our updated evidence synthesis. Our update confirms and extends the findings of the Climent-Sanz (2020)33 synthesis with nine additional studies, four of which evaluated the experiences of people with fibromyalgia who have used mind–body interventions for managing their fibromyalgia symptoms. Two further studies evaluated the experiences of fibromyalgia patients who attended group-based interventions for symptom management. Our update also provides a critical appraisal of the certainty of findings, which was not previously reported in the Climent-Sanz (2020) synthesis.33 While we have taken a systematic and rigorous methodological approach to this update of existing evidence, we acknowledge that qualitative interpretive approaches are subjective, and it is possible that different overall findings may have emerged had this update been conducted by other researchers. We broadened the inclusion criteria of the Climent-Sanz (2020)33 synthesis to consider adult and child populations and removed all language restrictions from our literature search. Unfortunately, we did not identify any eligible studies conducted in children. It is a limitation that the experiences of children who have fibromyalgia-related sleep problems are not captured in this update. We also did not identify any eligible studies published in non-English or non-Spanish languages. Although we employed hand-searching of the reference lists of included studies, we did not systematically search the grey literature. It is, therefore, unclear whether any relevant unpublished reports have been missed. Due to the limited reporting of participant demographic data by the authors of the included studies, it is unclear whether the study participants are representative of the wider population of fibromyalgia patients. Similarly, it was not possible to analyse data for diverse demographic groups, for example by race, ethnicity, or socioeconomic status, due to the way these data were reported by the study authors. We did not identify any eligible studies from low- or middle-income countries. It is therefore unclear whether people living with fibromyalgia in low resource settings have different experiences from people who live in high-income countries. Finally, one of the studies conducted by Colas and colleagues (2021) interviewed participants during the first COVID-19 national lockdown in France during April 2020 and May 2020.246 It is unclear whether the participants’ experiences of managing their fibromyalgia-related sleep problems were impacted differently during the lockdown period and the beginning of the COVID-19 pandemic, or to what extent they were impacted differently, than at any other point in time. It is, therefore, possible that the experiences of the participants in this study are less reflective of people living with fibromyalgia; however, we did not find any substantial differences in the findings for this study compared with the other studies included in our synthesis.

What this update adds to previous knowledge

By providing insights into people’s experiences of mind–body interventions and complex multidisciplinary group-based interventions to improve sleep, the findings of this evidence synthesis validate, confirm and extend the original findings published in 2020 by Climent-Sanz et al.33 Our updated synthesis also identified additional beliefs held by fibromyalgia patients about the causes of their poor sleep and highlighted the lack of data on the experiences of children and adolescents with fibromyalgia-related sleep problems and on adults from diverse and under-served groups. We also applied the GRADE-CERQual tool to the findings of our synthesis to provide an assessment of the confidence in the overall findings, which was not included in the original meta-synthesis. To assess the confidence in our overall findings, we applied the GRADE-CERQual tool to all qualitative studies, the newly identified studies and those included in the original meta-synthesis.