Journal of Psychosomatic Research 78 (2015) 420–426
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Journal of Psychosomatic Research
The most popular terms for medically unexplained symptoms: The views of CFS patients Federica Picariello a,b, Sheila Ali b, Rona Moss-Morris a, Trudie Chalder c,⁎ a b c
King's College London, United Kingdom South London and Maudsley NHS Foundation Trust, United Kingdom King's College London, United Kingdom
a r t i c l e
i n f o
Article history: Received 21 October 2014 Received in revised form 18 February 2015 Accepted 19 February 2015 Keywords: MUS CFS Terminology Labels Nomenclature Diagnosis Stigma Illness perceptions
a b s t r a c t Objective: Medically unexplained symptoms/syndromes are common, highly distressing and are often associated with profound disability. One of the controversies surrounding this area relates to which umbrella term should be used to group such symptoms. The purpose of this research was to establish the preferences of patients with chronic fatigue syndrome (CFS) for an umbrella term for medically unexplained symptoms. Methods: A cross-sectional mixed methods survey design was used. Participants were asked to indicate their three most preferred terms out of a list of commonly used terms and to provide any extra comments. Frequency analysis was employed to look at the preferences of terms for each rank. Comments were analysed using principles of inductive thematic analysis. Results: Eighty-seven patients with CFS completed a self-report survey. The term “Persistent Physical Symptoms” was the most popular first choice term chosen by 20.7% of patients. Terms containing the word “physical” were consistently more likely to be chosen. Three main themes emerged from the thematic analysis: 1) Physical nature of the illness, 2) Stigma, and 3) Evaluation of the terms, giving a more in-depth understanding of the findings. Conclusion: According to CFS patients, an umbrella term has to reflect the physical experience of MUS. © 2015 Elsevier Inc. All rights reserved.
Introduction Medically unexplained symptom (MUS) is the umbrella term applied to symptoms characterised by functional disability. These symptoms contribute to syndromes such as irritable bowel syndrome, chronic fatigue syndrome, fibromyalgia, non-cardiac chest pain and chronic tension headache. There is a high degree of overlap and the diagnostic label given to a particular patient may be influenced as much by the speciality in which the patient is seen as by the patient's symptoms [1]. A number of studies have shown that approximately half of new attenders (50%) to medical out-patient clinics have at least one medically unexplained symptom [2]. Patients with MUS incur twice the medical care utilisation and costs of other patients, independent of the presence of psychiatric and medical co-morbidity [3,4]. Left untreated the prognosis is poor [5,6]. Although the term MUS is commonly used in healthcare and research, many feel it is unsatisfactory for a range of reasons: 1) It makes an assumption that these symptoms might have a biomedical cause but fails to recognise that psycho-social causes are likely to be as ⁎ Corresponding author at: Department of Psychological Medicine, Institute of Psychiatry, Psychology & Neuroscience (IoPPN) King's College London, Weston Education Centre, 10 Cutcombe Road, London SE5 9RJ, United Kingdom. E-mail address: [email protected] (T. Chalder).
http://dx.doi.org/10.1016/j.jpsychores.2015.02.013 0022-3999/© 2015 Elsevier Inc. All rights reserved.
important if not more so, 2) Patients often feel their experience is invalidated as they perceive that the label implies that the symptoms are all in their mind and therefore not real, 3) It reinforces a dualistic philosophy of illness, and 4) The term fails to state what the problem is. Rather it states what it isn't [7,8]. Due to the heterogeneity of medically unexplained symptoms, it has been difficult to find a single universal term that can be used to refer to all of the conditions together. Many alternative terms have been suggested including bodily distress syndrome/disorder, and functional somatic syndromes. Bodily distress syndrome/disorder is a relatively new term that was recently introduced into the literature [9,10]. Creed et al [11] suggest that this term is suitable because it is accepted by both patients and healthcare professionals (HCPs), facilitates the possibility of multi-disciplinary treatment, is theoretically driven, does not reinforce dualism, and is neutral in relation to aetiology and pathology. However, some concerns arose in relation to the use of the word “distress”, as British participants were more likely than Danish and German participants to associate distress with a description of psychological states [11], suggesting that the meaning of a term can vary depending on language and cultural context. The term “Functional Somatic Syndrome” has historically been used in neurological settings [8]. It assumes that symptoms are related to an alteration of functioning, not structure [12]. According to Creed et al. [11], this term is acceptable to patients and health care professionals,
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as it is neutral with regard to the mind-body debate. It does not attempt to create a division between physical and psychological causes and does not rely on a single explanatory factor [13]. It also allows for flexibility for both medical and psychological treatments to be recommended to patients [8]. Labelling is potentially an important aspect of diagnosis. One purpose of it is to offer patients a legitimate “sick role”, and to validate the condition. The label confirms that the impairment is real and not imaginary [14]. Patients often gain relief when the various symptoms are given a label and this also unveils the different treatment options available to them [15]. However, the benefits of labelling have been questioned in relation to Medically Unexplained Symptoms, as the label might instead create more uncertainty and stigma [16]. A recent population study looked at the nomenclature preferences of 698 healthy adults. They found that the term “Persistent Physical Symptoms” was the top preference, followed by “Functional Symptoms” [17]. The aim of the current study was to find out which umbrella term patients with chronic fatigue syndrome (CFS) preferred, that could be used to describe not only their condition, but also illnesses such as IBS and non-cardiac chest pain. This study was intended to build on Marks and Hunter's study [17] using a population of patients with CFS in secondary care. Chronic Fatigue Syndrome, also known as myalgic encephalomyelitis (ME), is characterised by fatigue and numerous other symptoms, such as muscle pain and cognitive difficulties [18]. The illness has been estimated to affect at least 0.2–0.4% of the population worldwide [19,20]. The diagnosis of CFS can only be made after numerous tests have excluded medical or psychiatric causes for the symptoms [18,20]. The term CFS has triggered various debates and is often seen as misleading [21]. CFS patients frequently report that the term CFS trivialises the seriousness of this condition [18], as fatigue is only one of the many symptoms of this illness. Additionally, patients perceive that the consequences of fatigue are underestimated, because it is a common daily life experience of the healthy population [22]. In fact, CFS patients often display open disapproval towards psychological explanations of their illness [23]; therefore, this patient population may be more sensitive to a term used to describe conditions without a clear organic cause. Debates exist even in relation to the use of CFS versus ME [e.g. 21]. We were particularly interested therefore in which overarching term was preferred by patients with CFS. Method Design and procedure This study employed a cross-sectional mixed methods design. Participants were recruited using convenience sampling. Surveys were placed in the waiting room of an outpatient unit for CFS. Participants completed the survey while waiting for their appointment.
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Symptoms, 2) Functional Symptoms, 3) Functional Somatic Syndrome, 4) Functional Syndrome, 5) Bodily Distress Disorder, 6) Bodily Distress Syndrome, 7) Medically Unexplained Symptoms, 8) Medically Unexplained Physical Symptoms, 9) Persistent Physical Symptoms, 10) Somatoform Disorder, 11) No preference/don't know, and 12) Other. Participants were given space to add any further comments at the end of the first page. Participants were also asked to provide demographic information.
Analysis Patient preferences were analysed by looking at the frequency of each term being chosen. To assess the overall popularity of each term, each rank was assigned a score: 1) first choice was given three points, 2) second choice was assigned two points, and 3) third choice was assigned one point. Higher scores reflect a greater overall popularity across the ranks. The additional comments were coded by the identification of recurrent themes. Twenty-three participants out of 87 included additional comments. This qualitative analysis was guided by principles from inductive thematic analysis, a qualitative method used for “identifying, analysing and reporting patterns (themes) within data… without trying to fit it into a pre-existing coding frame, or the researcher's analytic preconceptions” [24]. All comments were analysed line-by-line and a final coding framework was devised (Appendix 2). In order to improve reliability, the data were coded by a second researcher and any discrepancies were discussed. The credibility of the analysis can be seen through a detailed paper trail in Appendix 1.
Results Sample Participants were included in the analysis if they self-reported being diagnosed with CFS and were eighteen years or older. Data was excluded from analysis if participants did not meet the above criteria and were carers, HCPs, not CFS patients, or did not provide this information. Eighty-seven participants were included in the final analysis. The sample was predominantly female (65.5%), of white ethnic origin (83.9%), with the majority falling in the age range of 25–54 (77.0%).
Main findings Table 1 shows the number (%) of patients who chose each term as their favourite for each rank: first, second, and third. The most popular first choice was Persistent Physical Symptoms (20.7%), and the second was Complex Physical Symptoms (17.2%). This popularity trend remained fairly consistent across the other rankings. Assigning scores to each term across the rankings, again revealed the same pattern (see Table 2). Terms including the word “physical” were most popular. Also, as can be seen from Fig. 1, there was a greater gap between the top two choices, and the rest of the terms.
Participants Participants were recruited from the Chronic Fatigue Syndrome Unit at the South London and Maudsley (SLaM) NHS Trust. This exploration of patients' views was considered an audit and approved by the South London and Maudsley NHS Foundation Trust and Psychological Medicine Clinical Academic Group. Materials Patients were asked to rank their top three preferred terms to describe conditions that are characterised by a variety of physical symptoms. Patients were asked: “If you had a physical symptom such as fatigue or pain that persisted and was found by doctors not to be caused by a particular disease, which of the following umbrella terms would you prefer most?”. The terms included in this survey were: 1) Complex Physical
Table 1 Popularity frequency of each term being chosen as first, second or third (N = 87).
Complex Physical Symptoms Functional Symptoms Functional Somatic Syndrome Functional Syndrome Bodily Distress Disorder Bodily Distress Syndrome Medically Unexplained Symptoms Medically Unexplained Physical Symptoms Persistent Physical Symptoms Somatoform Disorder No preference/Don't know Other
First choice
Second choice
Third choice
15 (17.2%) 4 (4.6%) 7 (8.0%) 6 (6.9%) 5 (5.7%) 3 (3.4%) 5 (5.7%) 10 (11.5%) 18 (20.7%) 4 (4.6%) 2 (2.3%) 8 (9.2%)
11 (12.6%) 3 (3.4%) 8 (9.2%) 3 (3.4%) 5 (5.7%) 4 (4.6%) 12 (13.8%) 8 (9.2%) 14 (16.1%) 5 (5.7%) 2 (2.3%) 1 (1.1%)
9 (10.3%) 8 (9.2%) 4 (4.6%) 5 (5.7%) 8 (9.2%) 4 (4.6%) 2 (2.3%) 6 (6.9%) 12 (13.8%) 2 (2.3%) 4 (4.6%) 0
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Table 2 Cumulative score for each term.
Participants unanimously agreed on the complex nature and consequences of CFS with its:
Term
Sum
Persistent Physical Symptoms Complex Physical Symptoms Medically Unexplained Physical Symptoms Medically Unexplained Symptoms Functional Somatic Syndrome Bodily Distress Disorder Functional Syndrome Functional Symptoms Other Somatoform Disorder Bodily Distress Syndrome No preference
94 76 52 41 41 33 29 26 26 24 21 14
“many different symptoms i.e. immune, skeletal, muscular, endocrine, adrenal, and neurological” (Unknown, 25–34).
Participants also suggested a number of additional terms that might be used. All included words such as “neurological” or “endocrine”, again emphasizing the biological attributions made by participants. Additionally, the majority of the suggested terms were classified as “syndromes” or “disorders”. For a full list of suggestions made by participants please see Appendix 3. 2. Stigma: This theme revolves around the treatment and support participants felt they had received from others. Many felt that HCPs had failed to validate the physical experience of MUS: “the way doctors discuss symptoms [can be] unsympathetic” (Female, 45–54).
Another participant, (Female, 55–64), seemed to portray a feeling of disillusionment with the medical profession: Thematic analysis
“The medical profession is letting us down.”
Three main themes emerged across the comments: 1) physical nature of the illness, 2) stigma, and 3) evaluation of the terms. 1. Physical nature of the illness: This theme reflects the importance placed by participants on the tangible nature of MUS. Participants' stated that these conditions are real, with physical symptoms and consequences, as well as a clear biological cause. Comments falling under this theme were the most commonly mentioned by the respondents, underlying the importance of a term being reflective of the physical reality of conditions such as CFS. This was one of the most prominent and consistent phrases that emerged throughout participants' narratives: “I think it is important the description acknowledges the affect it has physically and that it is not ‘imagined’” (Female, 25–34).
This relates back to theme 1, feeling that the physical reality of MUS is unrecognised. The possible strain that this can have on the patient-HCP relationship can be seen in the quote below: “Patients would rather doctors were honest and if they ‘don't know’ then say that rather than humiliating, frustrating and angering patients by subtly suggesting we seek psychiatric help…just because diagnostic tests (which doctors rely on too much) don't show anything to assist with diagnosis”(Female, 35–44).
This comment suggests that some HCPs may not effectively communicate the options available to patients with regard to treatment thereby creating a potential barrier between patients and HCPs. Another participant (Female, 45–54) conveyed a feeling of being misunderstood by the general public:
A number of participants explicitly used the words “imagined” and “real” (Unknown; Female, 45–54). Another participant (Unknown, 25–34) went further suggesting that science fails to validate the experience of MUS:
“It is essential for treatment development and research to increase awareness and understanding of symptoms throughout the general population.”
“‘Medically Unexplained Physical Symptom’: it doesn't matter to patients whether science can or cannot currently explain or understand the causes of the symptoms when it comes to naming them. They are real to the patient.”
3. Evaluation of the terms: Participants provided feedback on the concept of using an overarching label for conditions like CFS, and expressed their concerns about the appropriateness of specific words used within the different terms.
Other comments related to potential biological causes of CFS and participants expressed strong beliefs about the cause of CFS. This can be seen particularly well in the following quote: “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes” (Female, 55–64).
Umbrella term Some participants felt that an umbrella term would neither be useful nor appropriate (Female, 25–34; Female, 25–34; Female, 25–34), feeling that illnesses
Overall Popularity Score
120 100 80 60 40 20 0
Terms Used in the Survey
Fig. 1. Overall popularity bar chart.
F. Picariello et al. / Journal of Psychosomatic Research 78 (2015) 420–426 like CFS where no clear cause had been established were different both in manifestation and in aetiology: “I am not sure if an ‘umbrella’ term is particularly useful considering that IBS is very different to non-cardiac chest pain, for example. It [umbrella term] does not give an idea of where in the body the symptoms are” (Female, 25–34). Comments like this frequently emerged in support of this theme: “Ridiculous trying to lump the syndromes together” (Female, 25–34). This clearly demonstrates that a term needs to reflect the experience, nature and symptoms of an illness to be considered acceptable by patients. Underlying meaning Participants also provided feedback on the specific language used within the labels. Participants did not support the use of the word “syndrome”. There was both a lack of understanding as well as a concern that the word “implies [the illness is] bogus” (Female, 55– 64). Another word that was mentioned by two participants was “Distress” (Female, 25–34 & Female, 65–74). The participants suggested that this term was negative, subjective, and diverted attention from the physical suffering of patients, linking back to theme 1. Female (25–34): “‘Distress’ also sounds negative and may be alienating to those who feel too much emphasis is placed on their distress rather than the cause,” Female (25–34) expressed a view that words such as “distress” and “complex” were unnecessarily emotive and could have a negative connotation that could be stigmatizing for patients. This suggests that while a label should reflect transparently the experience of an illness it should not create fear of it.
Discussion Overview of findings The aim of this study was to identify CFS patients' preferences for terminology that could be used to group together varying medically unexplained conditions. These conditions are often referred to as medically unexplained symptoms because a biomedical cause has not currently been identified. The sample was predominantly female and between the ages of 25 and 54, reflective of the CFS patient population, and other MUS [2]. The findings from the survey showed that the most popular term was “Persistent Physical Symptoms”. The other most popular terms included the word “physical”, i.e. “Complex Physical Symptoms” and “Medically Unexplained Physical Symptoms”. The preference for terms including the word “physical” reflects the importance placed by the participants on the physical nature of such conditions. This is in line with previous findings, where MUS patients expressed dissatisfaction when diagnosed with incorrect psychiatric diagnoses [25]. As described by participants themselves, these symptoms are “real, and not imagined.” Despite the lack of a clear biological cause for CFS, patients will identify causes and will construct an illness identity. Contrary to the popularity ratings, where all the top choices were symptom-terms, some of the qualitative findings seem to suggest that participants liked terms classified as syndromes/disorders. Labelling symptoms and attributing symptoms to specific causes has consequences. One study which compared health outcomes of fatigued people who had made different attributions for their fatigue found that those who made physical attributions were less distressed at 18 months follow up but more disabled [26]. In another study, different terms were used for CFS, where the term ME triggered a more biomedical perception of the illness and patients were perceived as less likely to be fit for organ donation by undergraduate students and medical trainees [21]. Findings in context of previous studies Our results overlap somewhat with the findings of Marks and Hunter [17]. They found the term “Persistent Physical Symptoms” to be the top preference, chosen by 20% of the sample, while “Body Distress Disorder” was one of the least liked terms. It seems that a heterogeneous
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population sample have similar beliefs to a sample of patients with CFS. This contradicts the suggestion made by Creed et al. [11] that Body Distress Disorder was the more appropriate term. The use of the word “Distress” is likely to explain this disparity. However, it is important to acknowledge that there are consistent cross-cultural differences in the preferences for MUS terms, as has been observed in relation to the word “distress” [11]. For example, labels containing the word “functional” are considered offensive by many providers in the US [8], but are accepted in the UK by patients, but not all HCPs [8]. Consequently, cultural considerations are important, as the popularity ratings in this study are specific to a UK sample and might not hold true in other countries or cultures. The term “Complex Physical Symptoms” was more popular in our study than in the population study of Marks and Hunter [17]. However, some participants criticised the use of the word “complex” due to the attached negative connotation. Additionally, some participants questioned the utility of an umbrella term altogether to describe conditions that are symptomatically different. Even though, the most popular choices were all symptom-labels, the suggestions made by participants, in contrast, were mainly disorder or syndrome-labels. This finding can be explained by the stigma and lack of recognition often associated with MUS; therefore, disorder/syndrome labels may be more serious and legitimate in the eyes of patients. Another finding that emerged from the qualitative analysis was the consequence of a label in shaping HCPs and the general public's view of these conditions. Participants often felt a lack of empathy and understanding from HCPs. Indeed, a qualitative study comparing psychoeducation and Cognitive Behavioural Therapy (CBT) for CFS, found that validation was critical for patients, regardless of the effectiveness of treatment [27]. In line with the findings here, previous studies of CFS patients have consistently reported feeling blamed, dismissed and disbelieved by the medical profession [28,29]. In keeping with this perception fibromyalgia patients were prioritized and esteemed less by HCPs compared to conditions like myocardial infarctions, brain tumours and leukaemia [30]. Similarly, severity is also assumed from the type of treatment a patient is recommended. For example a CFS patient who is recommended a medical treatment is seen as being more disabled than someone who undergoes CBT with graded activity by HCPs [31]. This seems to suggest that the issues of recognition and stigma in the context of MUS are deep rooted. Consequently, changing a label might not necessarily translate into changes around the understanding and recognition offered by HCPs and others. Validation plays a crucial role in the diagnostic process, with studies reporting that as long as the reality of the symptoms is accepted, MUS patients will not reject psychological and social explanations [28,32]. Clinical implications There is a lack of knowledge and uncertainty associated with MUS amongst HCPs and the general public [e.g. 29]. This is illustrated in a study which reported that more than half of CFS patients were ill for more than three years before obtaining a diagnosis [15]. An appropriate label gives patients a language to discuss their complaints and experiences, as well as reassurance that there are other people suffering from the same physical symptoms [15]. A label can also help patients, HCPs and others to draw the distinction between the illness and the individual, contributing potentially to better outcomes and adjustment [15]. Nevertheless, the mere popularity of a term amongst patients is not sufficient to resolve the long-standing nomenclature debate in the context of MUS. Even though “Persistent Physical Symptoms” emerged as a favourite in this study, caution is warranted, as this term also has drawbacks. The term is vague and the nature of the problem is elusive. It fails to recognise that the symptoms may share a psychogenic aetiology and may apply to any illness or disease. In fact, as stated by Creed et al [11],
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no one term is perfect. Even within the debate between CFS versus ME, patients often prefer the label ME due to its biomedical implications. However, a ME diagnosis is often associated with a poorer prognosis [33,34]. Further research therefore is needed to understand the advantages and limitations of each term and to identify the most suitable term. It is important to acknowledge that merely changing labels cannot change the associated stigma and misunderstandings. Therefore, alongside identifying the most acceptable term, there is need to promote understanding and awareness about MUS amongst HCPs and the general public. Limitations A limitation of this study is the use of terms such as ‘somatoform’ that may require prior knowledge of medicine and therefore may not be understandable to all participants. Some participants commented that they were not familiar with some of the terms and this may have impacted on the choices they made in the survey. In addition, it may not be appropriate to compare certain umbrella terms, e.g. disorder or syndromes with symptoms. In addition, the findings are reflective of a CFS population attending secondary care. This does not necessarily generalise to MUS patients in primary care or other types of MUS. In fact, CFS patients are particularly sensitive to psychological explanations and nomenclature [21,23,33,34]. Additionally, participants were not approached to take part in this study, leading to a potential selection bias, where participants with strongest opinions might have chosen to fill out the survey. Another limitation of the study is the lack of ethnic diversity in the sample. Therefore, there may be limits to the generalizability of the results. Finally, it is important to note that qualitative analysis relies to some degree on the coder's understanding and interpretation, and potentially multiple ways of interpretation and construction of a coding manual are possible. To ensure that the qualitative analysis was reliable, two coders went through the data and edited the coding manual accordingly.
Appendix 1 Paper trail Stage 1 First, every interview was coded line-by-line.
Future directions There is a need for future research to explore whether an umbrella term is necessary at all and if so, what term would be most acceptable for patients, HCPs, and others. It may be useful to conduct semistructured interviews with CFS patients to explore the reasons behind the different choices and the acceptability of each term, obtaining richer qualitative data. It would also be useful to compare the preferences of people with different illnesses such as: IBS, or non-cardiac chest pain. Similarly, gathering the views and preferences of HCPs and carers of MUS patients, for example, via focus groups, could help to identify mismatches that may be impairing communication. Conclusion In conclusion, nomenclature is vital to appropriately reflect conditions, avoid misshaping opinions of HCPs and the public. According to CFS patients, a term has to reflect the physical experience of MUS. Further research is needed in order to explore what kind of terminology is best used when describing medically unexplained symptoms, therefore potentially increasing uptake and engagement with evidencebased treatments such as CBT. Declaration of interest To our knowledge there are no conflicts of interest to be declared. Acknowledgements We would like to thank the participants involved in this study and Caroline Foubister for her assistance with data collection. TC was in part supported by the NIHR Biomedical Research Centre for Mental Health at the South London and Maudsley NHS Foundation Trust and Institute of Psychiatry, King's College London.
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Stage 2 Comments were grouped together as part of a recurrent theme in every interview. Female, 55–64: 1. “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes.” 2. “Immune System Deficiency Chronic Fatigue (ISDCF)” Stage 3 Similar comments across the interviews were grouped together to identify patterns and links between the comments and possible subthemes were explored. Unknown, 25–34 “it should acknowledge the many different symptoms i.e. immune, skeletal, muscular, endocrine, adrenal, and neurological” Female, 55–64 “Endocrine, Adrenal and Cognitive Malfunction” Female, 35–44 “Neurological Lead Chronic Distress Syndrome” Male, 25–34 “‘ME’ vs. ‘CFS’ is still debated” Female, 55–64: “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes.” Stage 4 Comments of the kind across participants were then extracted into a separate word document and checked back with the narratives and a final theme was established: 1. physical nature of the illness. Theme
No
Description
Positive example
Negative example
Exemptions/Restrictions
Physical nature of the illness
1
Any mentions of the reality of the experienced symptoms and organic attributions.
“I think it is important the description acknowledges the affect it has physically and that it is not “imagined””. or “Chronic Fatigue is the result of a Virus lodging itself with the brain, causing immune system collapse
“‘Distress’ subjective as “mindfulness”, distinguishes between pain and suffering.”
Excluding any links to specific words or terms given in the survey
+ affects lymph nodes.”
Appendix 2 Coding manual. Theme
Sub-theme
No
Description
Positive example
Negative example
Physical nature of the illness
N/A
1
Any mentions of the reality of the experienced symptoms and organic attributions.
Excluding any links to specific “‘Distress’ subjective as words or terms given in the “mindfulness”, distinguishes between pain survey and suffering.”
Stigma
N/A
2
Comments in relation to the misunderstanding and negative perception displayed by others
Evaluation of the terms
Umbrella term
3.1 Evaluation of the usefulness of an umbrella term
“I think it is important the description acknowledges the affect it has physically and that it is not “imagined””. or “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes.” “the way doctors discuss symptoms [can be] unsympathetic.” and “It is essential for treatment development and research to increase awareness and understanding of symptoms throughout the general population” “Ridiculous trying to lump separate syndromes together”
Evaluation of the terms
Underlying meaning
3.2 Evaluation of the specific words used in terms
“‘Distress’ also sounds negative and may be alienating to those who feel too much emphasis is placed on their distress rather than the cause”
Exemptions/Restrictions
Excluding links to theme 1 in “I prefer terms which relation to the physical reality of acknowledge that a physical cause has not been suffering from CFS. found: this does not mean it does not exist.”
“I feel really strongly about this issue, as I believe ‘Chronic Fatigue Syndrome’ as it is, trivialises the condition.” “‘Medically Unexplained Physical Symptom’: it doesn't matter to patients whether science can or cannot currently explain or understand the causes of the symptoms.”
Excluding feedback on provided terms or CFS
Excluding feedback on the need for an umbrella term.
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Appendix 3 Label suggestions made by the participants. Female, 35–44 • Neurological Lead Chronic Distress Syndrome • Neurologically Lead Functional Distress Disorder • Persistent Neurological and Bodily Distress Syndrome • Persistent Neurological and Bodily Distress Disorder • Complex Functional Syndrome • Complex Functional Disorder Female, 55–64: • Chronic Fatigue • Somatic Dysfunction • Immune System Deficiency Chronic Fatigue (ISDCF) • Body Systems Dysfunction Unknown, 25–34 • Neuro-immune Disease Female, 55–64: • Body Energy Crisis Disorder • Endocrine, Adrenal and Cognitive Malfunction • Body Energy Crisis Malfunction • Non-rechargeable Body Energy Crisis Female, 35–44 • Diverse Medically Unexplained Symptoms Unknown, 25–34 • Complex Functional Disorder Syndrome
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[19] Wessely S, Chalder T, Hirsch S, Wallace P, Wright D. The prevalence and morbidity of chronic fatigue and chronic fatigue syndrome: a prospective primary care study. Am J Public Health 1997;87:1449–55. [20] Baker R, Shaw EJ. Diagnosis and management of chronic fatigue syndrome or myalgic encephalomyelitis (or encephalopathy): summary of NICE guidance. Br Med J 2007; 335:446. [21] Jason LA, Taylor RR, Stepanek Z, Plioplys S. Attitudes regarding chronic fatigue syndrome: the importance of a name. J Health Psychol 2001;6:61–71. [22] Taylor RR, Friedberg F, Jason LA. A clinician's guide to controversial illnesses: chronic fatigue syndrome, fibromyalgia, and multiple chemical sensitivities. Professional Resource Exchange Inc.; 2001 [23] Clements A, Sharpe M, Simkin S, Borrill J, Hawton K. Chronic fatigue syndrome: a qualitative investigation of patients' beliefs about the illness. J Psychosom Res 1997;42:615–24. [24] Braun V, Clarke V. Using thematic analysis in psychology. Qual Res Psychol 2006;3: 77–101. [25] Deale A, Wessely S. Patients' perceptions of medical care in chronic fatigue syndrome. Soc Sci Med 2001;52:1859–64. [26] Chalder T, Power MJ, Wessely S. Chronic fatigue in the community: ‘A question of attribution’. Psychol Med 1996;26:791–800. [27] Dennison L, Stanbrook R, Moss‐Morris R, Yardley L, Chalder T. Cognitive behavioural therapy and psycho‐education for chronic fatigue syndrome in young people: reflections from the families' perspective. Br J Health Psychol 2010;15:167–83. [28] Mayou R, Farmer A. Functional somatic symptoms and syndromes. Br Med J 2002; 325:265–8. [29] Webb CM, Collin SM, Deave T, Haig-Ferguson A, Spatz A, Crawley E. What stops children with a chronic illness accessing health care: a mixed methods study in children with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME). BMC Health Serv Res 2011;11:308. [30] Album D, Westin S. Do diseases have a prestige hierarchy? A survey among physicians and medical students. Soc Sci Med 2008;66:182–8. [31] Taylor RR, Jason LA, Kennedy CL, Friedberg F. Evaluating attributions for chronic fatigue syndrome based upon the type of treatment recommended. Rehabil Psychol 2001;46:165–77. [32] Salmon P, Dowrick CF, Ring A, Humphris GM. Voiced but unheard agendas: qualitative analysis of the psychosocial cues that patients with unexplained symptoms present to general practitioners. Br J Gen Pract 2004;54:171–6. [33] Hamilton WT, Gallagher AM, Thomas JM, White PD. The prognosis of different fatigue diagnostic labels: a longitudinal survey. Fam Pract 2005;22:383–8. [34] Huibers MJ, Wessely S. The act of diagnosis: pros and cons of labelling chronic fatigue syndrome. Psychol Med 2006;36:895–900.
Contents lists available at ScienceDirect
Journal of Psychosomatic Research
The most popular terms for medically unexplained symptoms: The views of CFS patients Federica Picariello a,b, Sheila Ali b, Rona Moss-Morris a, Trudie Chalder c,⁎ a b c
King's College London, United Kingdom South London and Maudsley NHS Foundation Trust, United Kingdom King's College London, United Kingdom
a r t i c l e
i n f o
Article history: Received 21 October 2014 Received in revised form 18 February 2015 Accepted 19 February 2015 Keywords: MUS CFS Terminology Labels Nomenclature Diagnosis Stigma Illness perceptions
a b s t r a c t Objective: Medically unexplained symptoms/syndromes are common, highly distressing and are often associated with profound disability. One of the controversies surrounding this area relates to which umbrella term should be used to group such symptoms. The purpose of this research was to establish the preferences of patients with chronic fatigue syndrome (CFS) for an umbrella term for medically unexplained symptoms. Methods: A cross-sectional mixed methods survey design was used. Participants were asked to indicate their three most preferred terms out of a list of commonly used terms and to provide any extra comments. Frequency analysis was employed to look at the preferences of terms for each rank. Comments were analysed using principles of inductive thematic analysis. Results: Eighty-seven patients with CFS completed a self-report survey. The term “Persistent Physical Symptoms” was the most popular first choice term chosen by 20.7% of patients. Terms containing the word “physical” were consistently more likely to be chosen. Three main themes emerged from the thematic analysis: 1) Physical nature of the illness, 2) Stigma, and 3) Evaluation of the terms, giving a more in-depth understanding of the findings. Conclusion: According to CFS patients, an umbrella term has to reflect the physical experience of MUS. © 2015 Elsevier Inc. All rights reserved.
Introduction Medically unexplained symptom (MUS) is the umbrella term applied to symptoms characterised by functional disability. These symptoms contribute to syndromes such as irritable bowel syndrome, chronic fatigue syndrome, fibromyalgia, non-cardiac chest pain and chronic tension headache. There is a high degree of overlap and the diagnostic label given to a particular patient may be influenced as much by the speciality in which the patient is seen as by the patient's symptoms [1]. A number of studies have shown that approximately half of new attenders (50%) to medical out-patient clinics have at least one medically unexplained symptom [2]. Patients with MUS incur twice the medical care utilisation and costs of other patients, independent of the presence of psychiatric and medical co-morbidity [3,4]. Left untreated the prognosis is poor [5,6]. Although the term MUS is commonly used in healthcare and research, many feel it is unsatisfactory for a range of reasons: 1) It makes an assumption that these symptoms might have a biomedical cause but fails to recognise that psycho-social causes are likely to be as ⁎ Corresponding author at: Department of Psychological Medicine, Institute of Psychiatry, Psychology & Neuroscience (IoPPN) King's College London, Weston Education Centre, 10 Cutcombe Road, London SE5 9RJ, United Kingdom. E-mail address: [email protected] (T. Chalder).
http://dx.doi.org/10.1016/j.jpsychores.2015.02.013 0022-3999/© 2015 Elsevier Inc. All rights reserved.
important if not more so, 2) Patients often feel their experience is invalidated as they perceive that the label implies that the symptoms are all in their mind and therefore not real, 3) It reinforces a dualistic philosophy of illness, and 4) The term fails to state what the problem is. Rather it states what it isn't [7,8]. Due to the heterogeneity of medically unexplained symptoms, it has been difficult to find a single universal term that can be used to refer to all of the conditions together. Many alternative terms have been suggested including bodily distress syndrome/disorder, and functional somatic syndromes. Bodily distress syndrome/disorder is a relatively new term that was recently introduced into the literature [9,10]. Creed et al [11] suggest that this term is suitable because it is accepted by both patients and healthcare professionals (HCPs), facilitates the possibility of multi-disciplinary treatment, is theoretically driven, does not reinforce dualism, and is neutral in relation to aetiology and pathology. However, some concerns arose in relation to the use of the word “distress”, as British participants were more likely than Danish and German participants to associate distress with a description of psychological states [11], suggesting that the meaning of a term can vary depending on language and cultural context. The term “Functional Somatic Syndrome” has historically been used in neurological settings [8]. It assumes that symptoms are related to an alteration of functioning, not structure [12]. According to Creed et al. [11], this term is acceptable to patients and health care professionals,
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as it is neutral with regard to the mind-body debate. It does not attempt to create a division between physical and psychological causes and does not rely on a single explanatory factor [13]. It also allows for flexibility for both medical and psychological treatments to be recommended to patients [8]. Labelling is potentially an important aspect of diagnosis. One purpose of it is to offer patients a legitimate “sick role”, and to validate the condition. The label confirms that the impairment is real and not imaginary [14]. Patients often gain relief when the various symptoms are given a label and this also unveils the different treatment options available to them [15]. However, the benefits of labelling have been questioned in relation to Medically Unexplained Symptoms, as the label might instead create more uncertainty and stigma [16]. A recent population study looked at the nomenclature preferences of 698 healthy adults. They found that the term “Persistent Physical Symptoms” was the top preference, followed by “Functional Symptoms” [17]. The aim of the current study was to find out which umbrella term patients with chronic fatigue syndrome (CFS) preferred, that could be used to describe not only their condition, but also illnesses such as IBS and non-cardiac chest pain. This study was intended to build on Marks and Hunter's study [17] using a population of patients with CFS in secondary care. Chronic Fatigue Syndrome, also known as myalgic encephalomyelitis (ME), is characterised by fatigue and numerous other symptoms, such as muscle pain and cognitive difficulties [18]. The illness has been estimated to affect at least 0.2–0.4% of the population worldwide [19,20]. The diagnosis of CFS can only be made after numerous tests have excluded medical or psychiatric causes for the symptoms [18,20]. The term CFS has triggered various debates and is often seen as misleading [21]. CFS patients frequently report that the term CFS trivialises the seriousness of this condition [18], as fatigue is only one of the many symptoms of this illness. Additionally, patients perceive that the consequences of fatigue are underestimated, because it is a common daily life experience of the healthy population [22]. In fact, CFS patients often display open disapproval towards psychological explanations of their illness [23]; therefore, this patient population may be more sensitive to a term used to describe conditions without a clear organic cause. Debates exist even in relation to the use of CFS versus ME [e.g. 21]. We were particularly interested therefore in which overarching term was preferred by patients with CFS. Method Design and procedure This study employed a cross-sectional mixed methods design. Participants were recruited using convenience sampling. Surveys were placed in the waiting room of an outpatient unit for CFS. Participants completed the survey while waiting for their appointment.
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Symptoms, 2) Functional Symptoms, 3) Functional Somatic Syndrome, 4) Functional Syndrome, 5) Bodily Distress Disorder, 6) Bodily Distress Syndrome, 7) Medically Unexplained Symptoms, 8) Medically Unexplained Physical Symptoms, 9) Persistent Physical Symptoms, 10) Somatoform Disorder, 11) No preference/don't know, and 12) Other. Participants were given space to add any further comments at the end of the first page. Participants were also asked to provide demographic information.
Analysis Patient preferences were analysed by looking at the frequency of each term being chosen. To assess the overall popularity of each term, each rank was assigned a score: 1) first choice was given three points, 2) second choice was assigned two points, and 3) third choice was assigned one point. Higher scores reflect a greater overall popularity across the ranks. The additional comments were coded by the identification of recurrent themes. Twenty-three participants out of 87 included additional comments. This qualitative analysis was guided by principles from inductive thematic analysis, a qualitative method used for “identifying, analysing and reporting patterns (themes) within data… without trying to fit it into a pre-existing coding frame, or the researcher's analytic preconceptions” [24]. All comments were analysed line-by-line and a final coding framework was devised (Appendix 2). In order to improve reliability, the data were coded by a second researcher and any discrepancies were discussed. The credibility of the analysis can be seen through a detailed paper trail in Appendix 1.
Results Sample Participants were included in the analysis if they self-reported being diagnosed with CFS and were eighteen years or older. Data was excluded from analysis if participants did not meet the above criteria and were carers, HCPs, not CFS patients, or did not provide this information. Eighty-seven participants were included in the final analysis. The sample was predominantly female (65.5%), of white ethnic origin (83.9%), with the majority falling in the age range of 25–54 (77.0%).
Main findings Table 1 shows the number (%) of patients who chose each term as their favourite for each rank: first, second, and third. The most popular first choice was Persistent Physical Symptoms (20.7%), and the second was Complex Physical Symptoms (17.2%). This popularity trend remained fairly consistent across the other rankings. Assigning scores to each term across the rankings, again revealed the same pattern (see Table 2). Terms including the word “physical” were most popular. Also, as can be seen from Fig. 1, there was a greater gap between the top two choices, and the rest of the terms.
Participants Participants were recruited from the Chronic Fatigue Syndrome Unit at the South London and Maudsley (SLaM) NHS Trust. This exploration of patients' views was considered an audit and approved by the South London and Maudsley NHS Foundation Trust and Psychological Medicine Clinical Academic Group. Materials Patients were asked to rank their top three preferred terms to describe conditions that are characterised by a variety of physical symptoms. Patients were asked: “If you had a physical symptom such as fatigue or pain that persisted and was found by doctors not to be caused by a particular disease, which of the following umbrella terms would you prefer most?”. The terms included in this survey were: 1) Complex Physical
Table 1 Popularity frequency of each term being chosen as first, second or third (N = 87).
Complex Physical Symptoms Functional Symptoms Functional Somatic Syndrome Functional Syndrome Bodily Distress Disorder Bodily Distress Syndrome Medically Unexplained Symptoms Medically Unexplained Physical Symptoms Persistent Physical Symptoms Somatoform Disorder No preference/Don't know Other
First choice
Second choice
Third choice
15 (17.2%) 4 (4.6%) 7 (8.0%) 6 (6.9%) 5 (5.7%) 3 (3.4%) 5 (5.7%) 10 (11.5%) 18 (20.7%) 4 (4.6%) 2 (2.3%) 8 (9.2%)
11 (12.6%) 3 (3.4%) 8 (9.2%) 3 (3.4%) 5 (5.7%) 4 (4.6%) 12 (13.8%) 8 (9.2%) 14 (16.1%) 5 (5.7%) 2 (2.3%) 1 (1.1%)
9 (10.3%) 8 (9.2%) 4 (4.6%) 5 (5.7%) 8 (9.2%) 4 (4.6%) 2 (2.3%) 6 (6.9%) 12 (13.8%) 2 (2.3%) 4 (4.6%) 0
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Table 2 Cumulative score for each term.
Participants unanimously agreed on the complex nature and consequences of CFS with its:
Term
Sum
Persistent Physical Symptoms Complex Physical Symptoms Medically Unexplained Physical Symptoms Medically Unexplained Symptoms Functional Somatic Syndrome Bodily Distress Disorder Functional Syndrome Functional Symptoms Other Somatoform Disorder Bodily Distress Syndrome No preference
94 76 52 41 41 33 29 26 26 24 21 14
“many different symptoms i.e. immune, skeletal, muscular, endocrine, adrenal, and neurological” (Unknown, 25–34).
Participants also suggested a number of additional terms that might be used. All included words such as “neurological” or “endocrine”, again emphasizing the biological attributions made by participants. Additionally, the majority of the suggested terms were classified as “syndromes” or “disorders”. For a full list of suggestions made by participants please see Appendix 3. 2. Stigma: This theme revolves around the treatment and support participants felt they had received from others. Many felt that HCPs had failed to validate the physical experience of MUS: “the way doctors discuss symptoms [can be] unsympathetic” (Female, 45–54).
Another participant, (Female, 55–64), seemed to portray a feeling of disillusionment with the medical profession: Thematic analysis
“The medical profession is letting us down.”
Three main themes emerged across the comments: 1) physical nature of the illness, 2) stigma, and 3) evaluation of the terms. 1. Physical nature of the illness: This theme reflects the importance placed by participants on the tangible nature of MUS. Participants' stated that these conditions are real, with physical symptoms and consequences, as well as a clear biological cause. Comments falling under this theme were the most commonly mentioned by the respondents, underlying the importance of a term being reflective of the physical reality of conditions such as CFS. This was one of the most prominent and consistent phrases that emerged throughout participants' narratives: “I think it is important the description acknowledges the affect it has physically and that it is not ‘imagined’” (Female, 25–34).
This relates back to theme 1, feeling that the physical reality of MUS is unrecognised. The possible strain that this can have on the patient-HCP relationship can be seen in the quote below: “Patients would rather doctors were honest and if they ‘don't know’ then say that rather than humiliating, frustrating and angering patients by subtly suggesting we seek psychiatric help…just because diagnostic tests (which doctors rely on too much) don't show anything to assist with diagnosis”(Female, 35–44).
This comment suggests that some HCPs may not effectively communicate the options available to patients with regard to treatment thereby creating a potential barrier between patients and HCPs. Another participant (Female, 45–54) conveyed a feeling of being misunderstood by the general public:
A number of participants explicitly used the words “imagined” and “real” (Unknown; Female, 45–54). Another participant (Unknown, 25–34) went further suggesting that science fails to validate the experience of MUS:
“It is essential for treatment development and research to increase awareness and understanding of symptoms throughout the general population.”
“‘Medically Unexplained Physical Symptom’: it doesn't matter to patients whether science can or cannot currently explain or understand the causes of the symptoms when it comes to naming them. They are real to the patient.”
3. Evaluation of the terms: Participants provided feedback on the concept of using an overarching label for conditions like CFS, and expressed their concerns about the appropriateness of specific words used within the different terms.
Other comments related to potential biological causes of CFS and participants expressed strong beliefs about the cause of CFS. This can be seen particularly well in the following quote: “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes” (Female, 55–64).
Umbrella term Some participants felt that an umbrella term would neither be useful nor appropriate (Female, 25–34; Female, 25–34; Female, 25–34), feeling that illnesses
Overall Popularity Score
120 100 80 60 40 20 0
Terms Used in the Survey
Fig. 1. Overall popularity bar chart.
F. Picariello et al. / Journal of Psychosomatic Research 78 (2015) 420–426 like CFS where no clear cause had been established were different both in manifestation and in aetiology: “I am not sure if an ‘umbrella’ term is particularly useful considering that IBS is very different to non-cardiac chest pain, for example. It [umbrella term] does not give an idea of where in the body the symptoms are” (Female, 25–34). Comments like this frequently emerged in support of this theme: “Ridiculous trying to lump the syndromes together” (Female, 25–34). This clearly demonstrates that a term needs to reflect the experience, nature and symptoms of an illness to be considered acceptable by patients. Underlying meaning Participants also provided feedback on the specific language used within the labels. Participants did not support the use of the word “syndrome”. There was both a lack of understanding as well as a concern that the word “implies [the illness is] bogus” (Female, 55– 64). Another word that was mentioned by two participants was “Distress” (Female, 25–34 & Female, 65–74). The participants suggested that this term was negative, subjective, and diverted attention from the physical suffering of patients, linking back to theme 1. Female (25–34): “‘Distress’ also sounds negative and may be alienating to those who feel too much emphasis is placed on their distress rather than the cause,” Female (25–34) expressed a view that words such as “distress” and “complex” were unnecessarily emotive and could have a negative connotation that could be stigmatizing for patients. This suggests that while a label should reflect transparently the experience of an illness it should not create fear of it.
Discussion Overview of findings The aim of this study was to identify CFS patients' preferences for terminology that could be used to group together varying medically unexplained conditions. These conditions are often referred to as medically unexplained symptoms because a biomedical cause has not currently been identified. The sample was predominantly female and between the ages of 25 and 54, reflective of the CFS patient population, and other MUS [2]. The findings from the survey showed that the most popular term was “Persistent Physical Symptoms”. The other most popular terms included the word “physical”, i.e. “Complex Physical Symptoms” and “Medically Unexplained Physical Symptoms”. The preference for terms including the word “physical” reflects the importance placed by the participants on the physical nature of such conditions. This is in line with previous findings, where MUS patients expressed dissatisfaction when diagnosed with incorrect psychiatric diagnoses [25]. As described by participants themselves, these symptoms are “real, and not imagined.” Despite the lack of a clear biological cause for CFS, patients will identify causes and will construct an illness identity. Contrary to the popularity ratings, where all the top choices were symptom-terms, some of the qualitative findings seem to suggest that participants liked terms classified as syndromes/disorders. Labelling symptoms and attributing symptoms to specific causes has consequences. One study which compared health outcomes of fatigued people who had made different attributions for their fatigue found that those who made physical attributions were less distressed at 18 months follow up but more disabled [26]. In another study, different terms were used for CFS, where the term ME triggered a more biomedical perception of the illness and patients were perceived as less likely to be fit for organ donation by undergraduate students and medical trainees [21]. Findings in context of previous studies Our results overlap somewhat with the findings of Marks and Hunter [17]. They found the term “Persistent Physical Symptoms” to be the top preference, chosen by 20% of the sample, while “Body Distress Disorder” was one of the least liked terms. It seems that a heterogeneous
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population sample have similar beliefs to a sample of patients with CFS. This contradicts the suggestion made by Creed et al. [11] that Body Distress Disorder was the more appropriate term. The use of the word “Distress” is likely to explain this disparity. However, it is important to acknowledge that there are consistent cross-cultural differences in the preferences for MUS terms, as has been observed in relation to the word “distress” [11]. For example, labels containing the word “functional” are considered offensive by many providers in the US [8], but are accepted in the UK by patients, but not all HCPs [8]. Consequently, cultural considerations are important, as the popularity ratings in this study are specific to a UK sample and might not hold true in other countries or cultures. The term “Complex Physical Symptoms” was more popular in our study than in the population study of Marks and Hunter [17]. However, some participants criticised the use of the word “complex” due to the attached negative connotation. Additionally, some participants questioned the utility of an umbrella term altogether to describe conditions that are symptomatically different. Even though, the most popular choices were all symptom-labels, the suggestions made by participants, in contrast, were mainly disorder or syndrome-labels. This finding can be explained by the stigma and lack of recognition often associated with MUS; therefore, disorder/syndrome labels may be more serious and legitimate in the eyes of patients. Another finding that emerged from the qualitative analysis was the consequence of a label in shaping HCPs and the general public's view of these conditions. Participants often felt a lack of empathy and understanding from HCPs. Indeed, a qualitative study comparing psychoeducation and Cognitive Behavioural Therapy (CBT) for CFS, found that validation was critical for patients, regardless of the effectiveness of treatment [27]. In line with the findings here, previous studies of CFS patients have consistently reported feeling blamed, dismissed and disbelieved by the medical profession [28,29]. In keeping with this perception fibromyalgia patients were prioritized and esteemed less by HCPs compared to conditions like myocardial infarctions, brain tumours and leukaemia [30]. Similarly, severity is also assumed from the type of treatment a patient is recommended. For example a CFS patient who is recommended a medical treatment is seen as being more disabled than someone who undergoes CBT with graded activity by HCPs [31]. This seems to suggest that the issues of recognition and stigma in the context of MUS are deep rooted. Consequently, changing a label might not necessarily translate into changes around the understanding and recognition offered by HCPs and others. Validation plays a crucial role in the diagnostic process, with studies reporting that as long as the reality of the symptoms is accepted, MUS patients will not reject psychological and social explanations [28,32]. Clinical implications There is a lack of knowledge and uncertainty associated with MUS amongst HCPs and the general public [e.g. 29]. This is illustrated in a study which reported that more than half of CFS patients were ill for more than three years before obtaining a diagnosis [15]. An appropriate label gives patients a language to discuss their complaints and experiences, as well as reassurance that there are other people suffering from the same physical symptoms [15]. A label can also help patients, HCPs and others to draw the distinction between the illness and the individual, contributing potentially to better outcomes and adjustment [15]. Nevertheless, the mere popularity of a term amongst patients is not sufficient to resolve the long-standing nomenclature debate in the context of MUS. Even though “Persistent Physical Symptoms” emerged as a favourite in this study, caution is warranted, as this term also has drawbacks. The term is vague and the nature of the problem is elusive. It fails to recognise that the symptoms may share a psychogenic aetiology and may apply to any illness or disease. In fact, as stated by Creed et al [11],
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no one term is perfect. Even within the debate between CFS versus ME, patients often prefer the label ME due to its biomedical implications. However, a ME diagnosis is often associated with a poorer prognosis [33,34]. Further research therefore is needed to understand the advantages and limitations of each term and to identify the most suitable term. It is important to acknowledge that merely changing labels cannot change the associated stigma and misunderstandings. Therefore, alongside identifying the most acceptable term, there is need to promote understanding and awareness about MUS amongst HCPs and the general public. Limitations A limitation of this study is the use of terms such as ‘somatoform’ that may require prior knowledge of medicine and therefore may not be understandable to all participants. Some participants commented that they were not familiar with some of the terms and this may have impacted on the choices they made in the survey. In addition, it may not be appropriate to compare certain umbrella terms, e.g. disorder or syndromes with symptoms. In addition, the findings are reflective of a CFS population attending secondary care. This does not necessarily generalise to MUS patients in primary care or other types of MUS. In fact, CFS patients are particularly sensitive to psychological explanations and nomenclature [21,23,33,34]. Additionally, participants were not approached to take part in this study, leading to a potential selection bias, where participants with strongest opinions might have chosen to fill out the survey. Another limitation of the study is the lack of ethnic diversity in the sample. Therefore, there may be limits to the generalizability of the results. Finally, it is important to note that qualitative analysis relies to some degree on the coder's understanding and interpretation, and potentially multiple ways of interpretation and construction of a coding manual are possible. To ensure that the qualitative analysis was reliable, two coders went through the data and edited the coding manual accordingly.
Appendix 1 Paper trail Stage 1 First, every interview was coded line-by-line.
Future directions There is a need for future research to explore whether an umbrella term is necessary at all and if so, what term would be most acceptable for patients, HCPs, and others. It may be useful to conduct semistructured interviews with CFS patients to explore the reasons behind the different choices and the acceptability of each term, obtaining richer qualitative data. It would also be useful to compare the preferences of people with different illnesses such as: IBS, or non-cardiac chest pain. Similarly, gathering the views and preferences of HCPs and carers of MUS patients, for example, via focus groups, could help to identify mismatches that may be impairing communication. Conclusion In conclusion, nomenclature is vital to appropriately reflect conditions, avoid misshaping opinions of HCPs and the public. According to CFS patients, a term has to reflect the physical experience of MUS. Further research is needed in order to explore what kind of terminology is best used when describing medically unexplained symptoms, therefore potentially increasing uptake and engagement with evidencebased treatments such as CBT. Declaration of interest To our knowledge there are no conflicts of interest to be declared. Acknowledgements We would like to thank the participants involved in this study and Caroline Foubister for her assistance with data collection. TC was in part supported by the NIHR Biomedical Research Centre for Mental Health at the South London and Maudsley NHS Foundation Trust and Institute of Psychiatry, King's College London.
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Stage 2 Comments were grouped together as part of a recurrent theme in every interview. Female, 55–64: 1. “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes.” 2. “Immune System Deficiency Chronic Fatigue (ISDCF)” Stage 3 Similar comments across the interviews were grouped together to identify patterns and links between the comments and possible subthemes were explored. Unknown, 25–34 “it should acknowledge the many different symptoms i.e. immune, skeletal, muscular, endocrine, adrenal, and neurological” Female, 55–64 “Endocrine, Adrenal and Cognitive Malfunction” Female, 35–44 “Neurological Lead Chronic Distress Syndrome” Male, 25–34 “‘ME’ vs. ‘CFS’ is still debated” Female, 55–64: “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes.” Stage 4 Comments of the kind across participants were then extracted into a separate word document and checked back with the narratives and a final theme was established: 1. physical nature of the illness. Theme
No
Description
Positive example
Negative example
Exemptions/Restrictions
Physical nature of the illness
1
Any mentions of the reality of the experienced symptoms and organic attributions.
“I think it is important the description acknowledges the affect it has physically and that it is not “imagined””. or “Chronic Fatigue is the result of a Virus lodging itself with the brain, causing immune system collapse
“‘Distress’ subjective as “mindfulness”, distinguishes between pain and suffering.”
Excluding any links to specific words or terms given in the survey
+ affects lymph nodes.”
Appendix 2 Coding manual. Theme
Sub-theme
No
Description
Positive example
Negative example
Physical nature of the illness
N/A
1
Any mentions of the reality of the experienced symptoms and organic attributions.
Excluding any links to specific “‘Distress’ subjective as words or terms given in the “mindfulness”, distinguishes between pain survey and suffering.”
Stigma
N/A
2
Comments in relation to the misunderstanding and negative perception displayed by others
Evaluation of the terms
Umbrella term
3.1 Evaluation of the usefulness of an umbrella term
“I think it is important the description acknowledges the affect it has physically and that it is not “imagined””. or “Chronic Fatigue is the result of a VIRUS lodging itself with the brain, causing immune system collapse + affects lymph nodes.” “the way doctors discuss symptoms [can be] unsympathetic.” and “It is essential for treatment development and research to increase awareness and understanding of symptoms throughout the general population” “Ridiculous trying to lump separate syndromes together”
Evaluation of the terms
Underlying meaning
3.2 Evaluation of the specific words used in terms
“‘Distress’ also sounds negative and may be alienating to those who feel too much emphasis is placed on their distress rather than the cause”
Exemptions/Restrictions
Excluding links to theme 1 in “I prefer terms which relation to the physical reality of acknowledge that a physical cause has not been suffering from CFS. found: this does not mean it does not exist.”
“I feel really strongly about this issue, as I believe ‘Chronic Fatigue Syndrome’ as it is, trivialises the condition.” “‘Medically Unexplained Physical Symptom’: it doesn't matter to patients whether science can or cannot currently explain or understand the causes of the symptoms.”
Excluding feedback on provided terms or CFS
Excluding feedback on the need for an umbrella term.
426
F. Picariello et al. / Journal of Psychosomatic Research 78 (2015) 420–426
Appendix 3 Label suggestions made by the participants. Female, 35–44 • Neurological Lead Chronic Distress Syndrome • Neurologically Lead Functional Distress Disorder • Persistent Neurological and Bodily Distress Syndrome • Persistent Neurological and Bodily Distress Disorder • Complex Functional Syndrome • Complex Functional Disorder Female, 55–64: • Chronic Fatigue • Somatic Dysfunction • Immune System Deficiency Chronic Fatigue (ISDCF) • Body Systems Dysfunction Unknown, 25–34 • Neuro-immune Disease Female, 55–64: • Body Energy Crisis Disorder • Endocrine, Adrenal and Cognitive Malfunction • Body Energy Crisis Malfunction • Non-rechargeable Body Energy Crisis Female, 35–44 • Diverse Medically Unexplained Symptoms Unknown, 25–34 • Complex Functional Disorder Syndrome
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