Overcoming the barriers to the diagnosis and management of chronic fatigue syndrome/ME in primary care: a meta synthesis of qualitative studies

What recommendations can be made for the development of the UK medical curriculum, the training of GPs and members of other health care professions, in order to overcome the barriers to the diagnosis and management of CFS/ME in primary care?

The focus of our work was based on two substantive areas. The first was the major themes and findings related to the barriers to diagnosis and management of CFS/ME, and the second was the implications and recommendations for overcoming these barriers. A literature search was performed using Medline/PubMed, PsycINFO, CINAHL and Web of Science databases. Our search keywords were Chronic Fatigue Syndrome or CFS or Myalgic Encephalomyelitis or Myalgic Encephalitis, which were combined, using Boolean logic terms “or” and “and”, with the following list of search terms: doctor; family physician; family practice; general practice; General Practitioner; GP; and Primary Care; service. We limited our search to published English language articles and to the last 30 years of publications. The search terms, inclusion criteria and the procedure for generation of the final sample of studies are displayed in Figure 1.

After removal of duplicates, the database searches produced 496 abstracts, dated from 1988 to 2013. All abstracts were screened using the following initial questions: Is this qualitative research?; Is this research based within a primary care setting?; Does it look at patient’s beliefs and views on the diagnosis, and management of CFS/ME?; Does it examine the practitioner’s perceptions of the diagnosis, management of CFS/ME?. This produced a subset of 47 articles which were reviewed by AW and AC. Consequently, 26 articles were excluded, which although they examined the process diagnosis and management of CFS/ME within primary care, they did not report the views of either patients or practitioner to this process. In total, 21 qualitative articles were included (Figure 1). Table 1 details the characteristics of these articles.

Guided by an existing quality appraisal framework that used a modified version of the CASP (Critical Appraisal Skills Programme) qualitative checklist, [38] a relevant quality appraisal tool was created by the authors and tailored to meet the aims of the meta synthesis (Appendix 1). Each paper was rated on the following criteria, using a three point scale (0 = Serious methodological issues; 1 = Minor methodological issues; 2 = Robust):

These ratings were then combined to categorise each paper as “key”, “adequate and relevant” or “flawed or not relevant” (Appendix 1). The aim was to remove any papers that were rated “flawed or not relevant” from our analysis.

Two researchers (AC and BF) carried out the quality appraisals. The researchers independently coded the remaining papers using the above criteria, thereby ensuring that all papers were double coded. Initial disagreements were successfully resolved via discussion and agreement was reached on all ratings. For example, it was highlighted that there was ambiguity around how to rate ‘relevance’ when a paper’s explicit research aim did not match the present study’s research question. Following discussion it was decided that if the findings explicitly identified ideas and concepts that answered the present study’s research question, that paper would not be penalised regarding its relevance. Furthermore it was decided that papers receiving the highest possible rating (2 for each criteria) would be categorised as key papers (Table 1). None of the 21 papers identified for analysis were classed as “not relevant”.

KB, AW and MG read and analysed each of the articles. A grid was constructed with studies along the x-axis and the content components (essential findings and interpretations) along the y-axis. Data was extracted on the original author’s analysis of the primary qualitative data. The themes derived from this primary data are called first order constructs [17, 18]. A thematic approach was then taken, grouping first order constructs from each paper into core themes. The researchers recorded which papers contributed to each theme, in terms of relevant data or contradictory or contrasting results. The analysis was completed independently and then the authors met to discuss, examine and agree on emergent themes. The final core themes are termed second-order constructs as they emerge from the analysis of first order analysis of the primary data. The recommendations for practice in this meta analysis are not only consistent with the original results but also extend beyond them, considering how the second order constructs sit within the wider literature. These recommendations for the medical curriculum are termed third order constructs [17, 18].

The literature shows that some health professionals have successfully diagnosed and managed CFS/ME by taking a more flexible, bio-psychosocial approach, building a positive, collaborative, therapeutic relationship with their patient. These lessons should inform the development of the medical curriculum in order to reduce inconsistencies in care.

In line with the findings of a review completed by Elliot [39] in the 1990s, our analysis suggests that the reluctance to diagnose and manage CFS/ME is based on scepticism and a lack of knowledge about the condition [7, 23, 26, 31, 40, 41]. Edwards et al. [28] reports that a lack of training at medical school on conditions commonly called “medically unexplained” can cause frustration in the patient which leads to a breakdown of the GP-patient relationship. Larun and Malterud [15] also highlight that a diagnosis and information on the condition was necessary for recovery, as without this, the patient can feel severely ill, yet blamed and dismissed and therefore disengage. This was also a theme in the current review which found that without a functioning relationship, communication breaks down and the GP is unable to learn from the patient’s experiences or form a therapeutic relationship to manage symptoms [31]. Patients are seen as difficult and un-cooperative which compounds the negative attitudes and stereotypical beliefs held by GPs about this group. Stenhoff et al., [12] describe how such negative beliefs are then passed on to medical students, creating a new generation of doctors with the same stereotypical views about patients with CFS/ME. This maintains the barriers to the diagnosis and management of CFS/ME that have existed for the past 20 years [12].

The barriers to the diagnosis and management of CFS/ME highlighted in this study result in a significant burden of dissatisfaction for both patients and health professionals [5, 24, 42, 43]. However, at least in the UK, the expectation that CFS/ME will normally be diagnosed and managed within primary care means that GPs need to find a way to engage with these patients [5].

This meta synthesis highlights that only a minority of health professionals report that they are able to successfully diagnose and manage patients with CFS/ME in primary care. This finding suggests that there is a need to address the inadequacy of medical training about this condition [5, 10, 15, 19, 23, 24, 31, 35]. For example, the somatisation model, used by many GPs to explain the symptoms of CFS/ME, arises out of the failure of the biomedical model [19, 32, 37]. It comes from reasoning that if there is no disease underlying the illness, the illness must be the manifestation of emotional distress. The bio-psychosocial model may be a better starting point for explaining and teaching medical students about conditions such as CFS/ME. This approach suggests a multi-factorial explanation for the condition, with interactions between biological, psychological and social factors maintaining symptoms. It focuses on a patient-centred consultation, using the collaborative and therapeutic skills shown to be valuable in this meta synthesis, to develop rapport and trust with a patient, listening and validating their experiences. Once the patient feels understood the GP can then go on to discuss the management of the condition [44]. CFS/ME therefore needs to be presented to students in a positive way, with clear and simple messages [45, 46]. This could mean teaching students about these conditions first before they start to habitually use the biomedical model as a prototype for all conditions. Stenhoff et al. [12] state that the first step in achieving this change is to address potential negative attitudes among trainers as sceptical attitudes towards CFS/ME can be learned by students.

An additional problem in the diagnosis and management of CFS/ME is the persistence of the term “medically unexplained illness”, which indicates diagnoses can only be by exclusion and does not allow for a positive diagnosis. This can provide a reason for medical professionals to delay diagnosis and even opt out of treatment as they feel that they have nothing to offer [47]. A fundamental shift in the perceived role of the GP is required to enable the holistic and therapeutic approach necessary to diagnose and manage CFS/ME in a positive way, as recommended by the NICE guideline [1].

The aim of this study was to systematically describe other interpretative studies. However, the varying research designs and level of detail provided in the qualitative studies (Table 1) make it difficult to objectively synthesise results across every study. The synthesis also only found studies from developed countries (UK, USA, Canada, Australia, Sweden and Norway), which used convenience sampling in primary care settings, CFS/ME support groups, and patient organizations. It is possible that there are other relevant studies that were not identified by our search terms. In order to address issues of consistency of reporting for this metasynthesis, the authors focused on peer reviewed, published papers only. The grey literature was therefore not included.

Formal validation of the novel quality appraisal tool used was not conducted which may have led to bias within quality coding judgements, However, double coding procedures were used to enhance the reliability of coder judgements thereby reducing any subjectivity which had potential to influence the results.

A number of the authors of this paper were involved in some of the original studies included in the synthesis. This is an inevitable finding from the limited number of researchers working in the field. In order to reduce bias, four researchers who were not authors of any of the original studies were involved in the reviewing, data extraction and analysis stages.