Patients’ experiences and effects of non-pharmacological treatment for myalgic encephalomyelitis/chronic fatigue syndrome – a scoping mixed methods review, by
Anne Marit Mengshoel, Ingrid Bergliot Helland, Mira Meeus, Jesus Castro-Marrero, Derek Pheby & Elin Bolle Strand in International Journal of Qualitative Studies on Health and Well-being Vol 15, 2020, no. 01
The EU COST Action 15111 collaboration on myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) aims to assess current research and identify knowledge gaps in Europe. Presently, our purpose is to map the effects of non-pharmacological therapies (NPTs) for ME/CFS, and what patients find important in the treatment process.
A scoping mixed methods literature review of European studies identified 16 papers fulfilling our inclusion criteria. The quantitative and qualitative studies were synthesized separately in tables. Additionally, extracts from the qualitative studies were subjected to translational analysis.
Effect studies addressed cognitive behavioural therapy (CBT, n = 4), multimodal rehabilitation (n = 2) and activity-pacing (n = 2). CBT reduced fatigue scores more than usual care or waiting list controls. The effects of rehabilitation and activity-pacing were inconsistent. The contents, assessment methods and effects of rehabilitation and activity pacing studies varied. For patients, health professionals’ recognition of ME/CFS and support were crucial, but they expressed ambiguous experiences of what the NPTs entail.
Methodological differences make comparisons across NPTs impossible, and from a patient perspective the relevance of the specific contents of NPTs are unclear. Future well-designed studies should focus on developing NPTs tailored to patients’ concerns and evaluation tools reflecting what is essential for patients.
The content and delivery of NPTs varied considerably in the studies, and any theoretical reasons for including the various components are not given. As NPTs are based on various theoretical assumptions, each NPT should first be tested to see if it is successful in achieving what it theoretically is supposed to do, and whether the changes are relevant in helping patients with ME/CFS. If successful in both respects, other components could be added step by step and tested in accordance with the framework of developing complex interventions (Craig et al., 2013; Richards & Hallberg, 2015). Future RCTs should also include long-time follow-up and cost-benefit analysis, and in particular, more studies on the role of NPTs for children and adolescents are needed.
The studies of CBT showed rather consistent results in having an effect on modifying fatigue, while the effects of activity pacing and multimodal rehabilitation were inconsistent. However, a timely question is whether the outcome measures were appropriate in evaluating whether the intentions of the NPTs, and what was found relevant for patients, were reached. At present, generic instruments, such as SF-36 and the CIS fatigue subscale, mostly assessed effects. A systematic literature review by Haywood et al. (2012) examined whether patient-reported measures, including those used in our review, had undergone rigorous, scientific evaluations in patients with ME/CFS. No evidence was found that patients had been involved in evaluating the relevance of any questionnaire, the content validities of which are therefore questionable. Likewise, measures showed little or moderate responsiveness to NPTs, which is essential in detecting effects. Another issue to be considered is how to interpret statistically significant differences in relation to clinical importance. It is questionable how much change is needed in an instrument’s scale to make it both clinically relevant and meaningful for patients (Angst et al., 2017). For future research, we recommend developing robust patient-reported disease-specific measures in collaboration with patients, and international consensus should be reached about the use of such instruments in making findings comparable across studies.
Patients with ME/CFS are not homogenous. Severely affected patients are house- or bedbound, while others, well represented in the effect studies we identified, struggle to maintain their living patterns. We did not find any studies on NPTs for severely affected patients. One reason for this may be that RCT designs are not appropriate in evaluating effects of such therapies for severely affected patients. To attempt to close this knowledge gap, case studies are high priority. Interviews with severely afflicted patients about their illness experiences, and interviews with patients that have become better after being bed- or house bond about what mattered for their progress, are needed to develop meaningful NPTs in the future. An alternative means of testing such new NPTs could be to apply single subject experimental designs in which a subject serves as his/her own control (Bates, 1996; Zhan & Ottenbacher, 2001). Patient-specific measures, such as Canadian Occupational Performance Measure as used by Kos et al. (Kos et al., 2015) can be appropriate to examine eventual progress but are less easy to use because of the semi-structured format. In terms of clinical practice, the semi-structured format has the advantage of being already a first step in the communication with the patients, but in research, reliability can be an issue when the assessors are not well trained.