‘A life I can cope with’. An alternative model of cognitive behavioural therapy (CBT) for CFS/ME

Abstract Objectives This study aimed to explore the experience of cognitive behavioural therapy (CBT) aimed at better management of chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME), rather than increasing activity. Design This was a qualitative study using grounded theory analysis. Methods Semi‐structured interviews were conducted with 13 adults who had engaged in CBT at a specialist CFS/ME service in which CBT is aimed at improved management of the condition. Results A model was produced in which participants felt more able to cope with CFS/ME. Reduced fatigue did not seem to be a necessary precondition to managing. This has implications for CBT for CFS/ME. Conclusions Specialist CBT for CFS/ME may result in improved coping and reduced distress, independently of changes in fatigue. Patient or Public Contribution The researcher met with a representative from the university's service user advisory group (SAGE), who had lived experience of CFS/ME. They commented on possible questions for the interview topic guide and provided advice on ways in which ethical issues specific to CFS/ME could be considered, for example, prevention of harm during interviews. Second, for quality assurance as part of respondent validation, a proposed draft of the grounded theory was discussed with participants.

states that CBT should also include more generic CBT components aimed at helping people adapt to illness, such as learning assertiveness skills to help people set limits on their activity, and supporting adjustment to the diagnosis. The recent 2020 draft National Institute for Health and Care Excellence (NICE) guidance represents a shift from the 2007 guidance, 1 highlighting that CBT is not a treatment or cure for ME/CFS, but may be useful in supporting people who live with ME/CFS to manage their symptoms. 2 A Cochrane meta-analysis 3 reported that CBT was more effective than usual care and other psychological therapies for reducing fatigue in adults with CFS/ME, with 40% of participants showing improvement after CBT compared with 26% in usual care. However, a large patient survey conducted by the UK's M.E. Association found that between 8% and 35% of participants reported improvement after CBT and 18% reported adverse effects. 4 Yet, there has been considerable publicity on how effective CBT is for CFS/ME. Friedberg 5 suggests that this may underpin the considerable backlash against CBT from patients, much of which has been on social media.
The new draft NICE Guidance 2 recommends CBT as a form of support for self-management of CFS/ME, but does not suggest that it will eliminate difficulties.
It has been highlighted that the fundamental treatment goals for CBT for CFS/ME vary across studies. 6 Some CBT interventions, such as those investigated in the high-profile PACE trial, 7 adopt a model that aims to increase activity to promote recovery. This is based on the 'cognitive behavioural model of CFS', 8 which suggests that CFS/ ME is maintained by unhelpful beliefs about exercise and illness, for example, 'This activity will make me feel worse' (p. 537), which result in activity avoidance and subsequent changes in physiology such as loss of muscle strength, collectively known as 'deconditioning'. The cognitive theory suggests that reconditioning can lead to recovery from the condition. Another model of CBT for CFS/ME aims to instead support people to adapt to the condition and find an optimal level of activity to minimise exacerbation in symptoms. 6 Most CFS/ ME patients do not believe that their illness is psychogenic and can be cured by increasing their activity, although many acknowledge that movement may play some role in recovery. 9,10 The M.E. Association has advocated that interventions focused on better management of the condition are more useful for CFS/ME than interventions whose primary goal is an increase in activity. Yet, despite this, many people with CFS/ME are offered CBT focused primarily on increasing activity. 10 NICE 1 highlights that one aim of CBT should be to reduce the 'distress associated with CFS/ME'. Yet, CFS/ME intervention studies have typically focused on fatigue and functioning as primary outcome measures, rather than changes in distress. The available evidence on psychological outcomes for CBT for CFS/ME is mixed. Price et al. 3 found that CBT failed to reduce distress in comparison to usual care.
However, a meta-analysis by Castell et al. 11 showed that following CBT, participants experienced a significantly greater reduction in depression and anxiety than controls. As yet, there are no studies aimed at understanding processes via which a reduction in psychological distress may follow from CBT for CFS/ME. Brooks et al. 12 found that after CBT, patients showed significantly increased acceptance of the condition and reductions in two aspects of perfectionism (concern over mistakes and doubts about actions). It was found that increased acceptance post-CBT was significantly correlated with improvement in fatigue, physical functioning and work and social adjustment.
However, research exploring psychological change following CBT for CFS/ME is in its infancy and the specific perceived relationships between coping, acceptance, fatigue and other factors are yet to be explored. In addition, there are very few studies exploring models of CBT with the primary goal of supporting participants to live with CFS/ME, rather than increasing their activity to reduce fatigue. Yet, this former model is being offered by services and warrants further investigation.
This study therefore sought to build a preliminary model of therapeutic change for CBT aimed at better adjustment to and management of CFS/ME, exploring the inner workings of the model and ways in which it may effect change.

| Design
This study was qualitative and used interviews.

| Participants
Participants (n = 13) had all been assessed as fulfilling the criteria for a diagnosis of CFS or ME by a doctor in the service. The service adopted a pragmatic approach to diagnosis, in which clinicians used more than one set of diagnostic criteria. The median age of the participants was 49 (range: 19-72). As shown in Table 1, six participants were in paid work, one in education, five were retired or unemployed and one was a home-maker. Participants reported attending between 6 and 10 sessions of specialist CBT at the service (median = 6). Three participants had previously undertaken additional episodes of specialist CBT for CFS/ME at other services. They reported that, as within the service, the goal had been positioned as better management of CFS/ME; therefore, data relating to these experiences were included in the analysis. Change scores were not noted for this study, but participants mainly reported lack of change on physical symptoms. The lack of questionnaire data is revisited in Section 4.

| Procedure
Participants were recruited through an NHS specialist community CFS/ME service. In contrast to the CBT models adopted in many CFS/ ME services, 11 the model of CBT delivered in this service was not one with the primary aim of increasing activity to facilitate 'reconditioning' and a reduction in fatigue. Instead, the primary goal was increased quality of life by better adjustment to and management of CFS/ME. The therapists did not follow a manual or use a specific model of CBT for CFS/ME, but rather drew on a range of CBT techniques and concepts, both specific to CFS/ME (e.g., pacing) and more general (e.g., lowering standards, assertiveness, self-soothing), tailoring the therapy to each individual. A reduction in fatigue was an anticipated secondary benefit for the intervention. CBT was delivered by two CBT therapists in the service who had been qualified for 7 and 11 years, respectively.
Following ethical approval, the service contacted all service users who had completed CBT within the previous 2 years about the study by letter. Participants were given an information sheet containing information about confidentiality, risk and the right to withdraw from the study at any time. Individual semi-structured interviews lasted between 52 and 93 min. Six interviews were conducted in person in a research space adjacent to the service and seven were conducted over Skype. All interviews were conducted by the first author, a female trainee clinical psychologist aged between 30 and 40 years with previous experience of conducting research interviews at the doctoral level. The interviewer was not associated with the service and was not known to the participants. Interviews were audio-recorded and transcribed. Analysis was primarily conducted by the first author.

| Data analysis
Analysis began after the first interview and continued concurrently with data collection. It was informed by the approach described by Strauss and Corbin. 13 Constant comparison and memoing were used throughout to identify and record similarities and differences between data from different participants and to aid category development. Theoretical sampling 14 was achieved by adapting interview questions according to emerging categories within the data.
Analysis began with detailed line-by-line open coding aimed at identifying concepts within the data. It then progressed to axial coding, exploring the properties and dimensions of categories. Data were then analysed for context, keeping in mind the concept of conditional/ consequential matrices. 13 Relationships between categories were explored by creating maps of each participant's experience. The later stages of analysis involved selective coding and broader theoretical integration. In the final stages, constant comparison highlighted that new data reflected existing major categories in the model and that these categories were dense in terms of properties and well integrated, suggesting theoretical saturation. 13 Dimensional variation was more limited due to the absence of participants who reported poorer outcomes from the intervention.

| Quality assurance
Elliott et al's. 15 quality guidelines were used to increase the credibility of the research. A research diary was maintained, a bracketing interview was conducted and a positioning statement was created.
Two line-by-line coded transcripts, memos around emerging categories and integrative maps were shared with the second author, who offered comments, challenges and elaborations, which were discussed and incorporated into the analysis. The second author, like the first, had no connection with the service. Finally, respondent validation was undertaken to ascertain the credibility of the analysis to participants. 16 A proposed draft of the theory was sent to participants and discussed via phone or email.

| RESULTS
As shown in Table 2, 10 main categories were developed from the data. The categories are illustrated in the model in Figure 1. The principal context of the therapy experience was dealing with ongoing symptoms. Four categories describe experiences in the therapy room (illustrated in the top circle in Figure 1) and three categories describe experiences in daily life (illustrated in the bottom circle in Figure 1). As illustrated by the shaded circles in Figure 1, there was often interaction between the different experiences within each context. Experiences in the therapy room and daily life seemed to lead to changes in the meaning that participants held around their lives with CFS/ME, as reflected in the categories 'A life I can cope with' and 'Accepting the reality of CFS/ME'. Each of the categories and their subcategories will be discussed, with emphasis on the relationships between them.
Participants have been assigned pseudonyms to maintain anonymity.

Context Category Subcategory
Not specific to one context

| Dealing with ongoing symptoms
In the first of two subcategories, participants described lack of improvement from prior interventions. Despite engaging in a range of interventions before CBT, all but one participant continued to experience significant CFS/ME symptoms. Jean was an exception as she felt '85% back to normal' when starting specialist CBT, which she attributed to graded exercise: The weird thing about ME is that the only cure seems to be, you know, the graded exercise. (Jean) Participants also described experiencing minimal changes over CBT: Most participants experienced no change in their CFS/ME Making progress seemed more likely when participants felt that the therapist adopted a holistic approach and understood their unique difficulties: It wasn't just focused on ME. It was looking at my whole body or mind and seeing how I was coping with things, so I was actually really pleased. (Susan) Participants were consistently told that the goal of therapy was better management of CFS/ME. All participants found this suitable as it fitted with their experience of the condition.

| Seeing myself and CFS/ME differently
There were four subcategories, first: CFS/ME is real and not my fault.
For several participants, therapists highlighting that CFS/ME was a real and chronic condition gave participants a clearer representation of the illness and its impact: In the present study, participants felt more in control and saw it as more possible to live alongside the condition. Partly, this seemed to result from addressing issues that were preventing participants from being able to manage their CFS/ME, such as holding themselves to high standards. This echoes Brooks et al., 12 who found reduced perfectionism and fatigue after CBT for CFS/ME. Brooks et al. 12 questioned whether the reduction in perfectionism in fact reflected a reduction in 'goal discrepancy' (being unable to achieve a valued goal) as a knock-on effect of reduced fatigue. The present study suggests otherwise, as participants described lowering expectations of themselves, often without any associated improvement in fatigue. In fact, data suggested that it was a lowering of standards that led to better management of symptoms, both directly and via increased acceptance of the condition.
This, together with learning tools such as problem solving and pacing, seemed to allow participants to approach their daily lives in a more adaptive way, preventing symptom flare-ups and allowing more time and energy for valued activities. Participants valuing tools for managing energy or 'pacing' echoes the survey results presented in Geraghty et al. 4 Participants felt more able to manage their daily lives after learning tools for managing emotions and resolving sources of distress in their broader lives, such as traumas or social anxiety.
Participants felt more able to cope with CFS/ME after addressing anxieties about how CFS/ME prevented them from being able to fulfil their roles or values. Thus, as aimed for in CBT, results suggested a shift in both cognitions and behaviours. These were not, however, those cognitions and behaviours cited as key targets in the dominant cognitive behavioural models of CFS 7,8 that is, unhelpful illness beliefs and activity avoidance. CBT is an umbrella term, and our results confirm that multiple models of CBT are in use within the field of CFS/ME.

| Acceptance
In line with studies of other psychosocial interventions for CFS/ME, [19][20][21] including CBT, 12  Lastly, in terms of replicability, the therapists at the service were not able to provide a therapy protocol. They described general issues often addressed in the therapy (e.g., lowering high standards, pacing), but were guided by clinical judgement to individualise therapy for each client. For this reason, it may be important in future studies to incorporate in-depth interviews with therapists and video-assisted analysis of therapy to more clearly understand therapeutic processes and link these with client-reported experiences and outcomes, such as those described in this study.

| Clinical implications
This study suggests explanations as to how CBT with the primary aim of improving quality of life rather than increasing activity and redu- approach, which allowed them to address a wide range of issues in their lives preventing them from accommodating and adapting to CFS/ME. Some components of the model go even broader than CBT and are more features of supportive therapies such as counselling (offering hope and a space to 'lighten the load'). It is useful to note that for common mental health difficulties, it has been found that therapeutic alliance and therapist characteristics are key predictors of outcomes across client groups, for example see Budd and Hughes. 23 Both of these may be important factors in this study, in which experienced therapists were adapting treatment to each participant. It is important to note that all participants in this study found it appropriate that the goal of therapy was positioned as better management of CFS/ME and quality of life rather than physical recovery. This model of CBT may not be suitable for service users who view the goal as recovery of pre-CFS/ME activity levels.

| Directions for future research
Research exploring psychological outcomes and processes in CBT for CFS/ME, aimed at better adapting to the illness rather than with the primary aim of increasing activity, is in its infancy. It may be appropriate to explore aspects of the model developed here through quantitative research: for instance, measuring changes in distress, acceptance and quality of life at intervals during this model of therapy. Mediation analyses might include self-efficacy, sense of control, self-compassion, perfectionism, 'goal discrepancy' and acceptance or denial of having a chronic illness or condition. It may also be useful to identify the active components of therapy using component analysis. 24 Several theories implicate the body's stress hormonal systems as the basis of CFS/ME; it has been suggested for example that the condition arises from the same physiological processes as those that occur in 'burnout'. 25 It could therefore be hypothesised that the improvement in well-being found in this study could lead to a reduction in fatigue in the long term. This requires further research.
Multiple case research might examine how apparent improvements in daily living may lead to less experience of fatigue and activity restriction, and the points at which small successes and setbacks occur during the course of CBT, as well as outside of therapy.
Further research might also consider whether the current model could be extended to service users with more severe fatigue, for example, those who are homebound.

| CONCLUSION
The study produced several novel findings. First, CBT aimed at improved management of CFS/ME, rather than increased activity, was viewed as acceptable by participants and appeared to alleviate distress about living with the condition. Second, this was not dependent on improvements in fatigue, and seemed to be facilitated by exploring and resolving issues around standards and identity and learning tools such as problem solving and pacing. Together, these seemed to facilitate participants adopting a more adaptive approach to daily life and becoming able to do more of what they valued.
A common part of the therapeutic process was increased acceptance of the reality of having CFS/ME, leading to more adaptive coping.
The results support the need for interventions addressing the above factors. However, it is unclear to what extent these apparent CLARK AND HOLTTUM | 101 improvements in daily living may be maintained in the long term, and whether they may lead to less experience of fatigue and activity restriction. Further research is needed to test out the model suggested in this study.