Family Practice Advance Access published online on July 1, 2008
Family Practice, doi:10.1093/fampra/cmn041
Medically unexplained symptoms in family medicine: defining a research agenda. Proceedings from WONCA 2007
a Department of Family Medicine, Radboud University Medical Centre, Nijmegen, The Netherlands
b Divison of Primary Care, University of Liverpool, Liverpool, UK
c Medical Psychology and Mental Health Unit, Medical Sciences Faculty, State University of Rio de Janeiro, Rio de Janeiro, Brazil
d Family Medicine Unit, The University of Hong Kong, Hong Kong SAR, China and
e Department of General Practice, VU University Medical Center Amsterdam, Amsterdam, The Netherlands. Correspondence to TC olde Hartman, 117 Department of General Practice/Family Medicine, Radboud University Medical Centre, PO Box 9101, 6500 HB Nijmegen, The Netherlands; Email: t.oldehartman{at}hag.umcn.nl
Received 27 September 2007; Revised 5 June 2008; Accepted 6 June 2008.
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Abstract |
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Background. Medically unexplained symptoms (MUSs) are frequently presented in primary care. Unfortunately, knowledge of these patients and/or symptoms in primary care is still limited. Available research comes mainly from Europe and North America, while the perspectives of cultures such as Africa, Asia and South America are relatively unknown. To bring cultural perspectives together, a symposium and workshop on MUS in primary care was held at the WONCA World Conference 2007 in Singapore.
Objective. Main goals of this symposium and workshop—apart from presenting ongoing research and bringing together experts in MUS—were to detect knowledge gaps in MUS and to establish priorities in MUS research. This publication focuses on the proposed research agenda.
Methods. Using a nominal group technique, we generated research topics and set priorities. Research topics were grouped into research themes.
Results. Participants’ (66 researchers and GPs from 29 nationalities) most important research topics were ‘formulating a broadly accepted definition of MUS’, ‘finding a strategy to recognize MUS better and at an earlier stage’, ‘studying the value of self-management and empowerment in patients with MUS’ and ‘finding predictors to decide which strategy will best help the individual patient with MUS’. Priorities in research themes of MUS are as follows: (i) therapeutic options for patients with MUS and (ii) problems in consultations with these patients.
Conclusions. More research on MUS in primary care is needed to improve the consultations with and management of these patients. Internationally primary care conferences are excellent for exchanging ideas and formulating central issues of research.
Keywords. Family practice, medically unexplained symptoms, research agenda, cultural perspective.
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Introduction |
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Acute medically unexplained symptoms (MUSs) are common in family medicine. Twenty to fifty per cent of all contacts between patient and GPs concern unexplained physical symptoms.1,2 Mostly, these symptoms resolve or do not need further medical attention after one consultation. However, there is a group of patients who frequently consult the GP with unexplained symptoms. In a Dutch survey of GP–patient contacts, the prevalence of patients with at least four contacts per year for MUS was 2.5%.3 Frequent attenders with MUS have a high risk of extensive investigations and referrals.4 Only about a quarter of patients frequently presenting MUS met the criteria of one of the DSM-IV somatoform categories (i.e. somatization or undifferentiated somatoform disorder).5 This means that the majority of patients with MUS in primary care are different from patients meeting DSM-IV criteria for somatoform disorder. Consequently, this relevant group of patients in primary care deserves to be studied seperately.6 However, study and hence knowledge of many aspects of this group of MUS patients is scarce. For instance, we are still debating the exact definition and classification of MUS which is strongly related to cultural differences.7 Then, we know little about the prognosis of the symptoms, and we have limited knowledge about factors predicting the prognosis or its course of MUS over time. Furthermore, there is doubt about the best approach to and treatment of these patients.
In the traditional approach of patients who repeatedly present MUS, GPs try to reassure them by giving some—but often inadequate—explanation about the symptoms. Furthermore, concerns of patients with MUS are less likely to be explored and facilitation of patients’ talk about psychosocial problems is limited.8–10 As many patients (and doctors) are not satisfied by this traditional approach, in recent years some research has been done on cognitive behavioural therapy (CBT)-based interventions. These studies show conflicting results. Most studies show no effect of CBT interventions on well-being, sick leave and limitations due to the symptoms.11–14 Several explanations may be offered for these disappointing results: the training of the GPs might have been insufficient or important issues such as the anxiety of the patients might not have been adequately addressed. Furthermore, an adequate explanation of the symptoms and non-specific effects caused by mutual trust, empathy and shared understanding have not been studied.15 Alternatively, one might ask whether the outcomes, such as number and duration of symptoms, well-being, sick leave and limitations that doctors or researchers usually study, are the outcomes that patients would also prefer. We must conclude that there are still many uncertainties about this group of patients.
Although MUS is a universally common phenomenon, most research on MUS comes from Europe and North America. The perspectives of other cultures such as Africa, the Asia Pacific and South America are relatively unknown. It is known from recent research that there are cultural differences in the distribution of MUS and the meaning and significance of a symptom depends on the perceived relationship with diseases in a culture.7,16 Moreover, cultural and social norms determine whether a person with a particular MUS can adopt the sick role: cultures such as the Chinese reject psychological symptoms and therefore cause psychosomatization.17 Bringing these international perspectives together might help to fill the gaps in knowledge about MUS.
We therefore organized a symposium and a workshop about MUS in primary care during the WONCA World Conference 2007 (Singapore, July 26). Our aim was to gather topics for research on MUS from discussions with researchers and GPs from all over the world and to prioritize these topics in order to establish a global research agenda.
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Methods |
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During the symposium ‘MUS in family medicine: the state of the art’, GP researchers presented ongoing research in MUS in different countries all over the world. The two main themes were the classification and cultural variation of MUS and the process of consultation with MUS patients.18–20 The symposium was also meant as an introduction to the subsequent workshop. The aim of this workshop ‘MUS in family medicine: where should we go?’ was to bring together GPs with a broad cultural variety interested in MUS to discuss ideas about research topics in the field of MUS and to make choices on which ideas needed priority.
Participants in the workshop
In order to discuss the various perspectives of MUS research and bring together GPs from different cultural backgrounds to look at the broader picture of MUS, we invited researchers and interested GPs from different nationalities. The nationality of each participant in the workshop was registered by one of the organizers (TCoH).
Nominal group technique
In the workshop, we used the nominal group technique to generate a research agenda. In the nominal group technique, participants are brought together for discussion. The procedure usually comprises four stages: generating ideas, recording of those ideas, clarification and priority voting.21 This method encourages contributions from everyone by allowing equal participation among participants. Moreover, it is a structured, transparent and replicable way of synthesizing and generating ideas.22
During the workshop, we generated ideas by stimulating discussion: we allowed 10 minutes of brainstorming in small groups to discuss what they thought to be the most relevant MUS research topics. Each small group made a shortlist of their most important research topics. We continued with an inventory of all relevant topics arising from these discussions. We wrote the topics onto flip charts. If necessary, the discussion leaders (LJAH-F, EmvW-B) asked for clarification. After this inventory, each participant was asked to choose five topics with the highest priority from this list and rank them. Finally, we asked all the participants to vote, allocating the highest score (5) to the most important research topic, then 4 to the next most important statement and so on, by sticking coloured cards on the flip charts. The organizers of the workshop and preceding symposium circulated the room during this task, giving assistance where needed.
The research topics with the highest votes were presented to the audience and discussed briefly to clarify possible research methodologies suitable to answer the research question.
Data analysis
One of the researchers (TCoH) summed the votes for each research topic that was brought up. According to the voting scores, topics were ranked, producing a list of the participants’ ‘priority research topics’. Identical scores were given the same ranking number; research topics with higher number of individual votes were listed higher.23 Finally, research topics were grouped into research themes.
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Results |
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Baseline characteristics of workshop attendees
A total of 66 researchers and GPs from 29 nationalities from 6 different continents attended our workshop (Table 1). Fourteen participants (21%) only attended the introduction of the workshop.
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Most important topics in MUS research
The nominal group technique resulted in a total of 29 research topics. In Table 2, the ranking of these research topics is shown.
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Almost half (48%) of the audience agreed that the most important problem is that MUSs are not clearly defined. There is need for an unambiguous definition of MUS that can be applied globally. Without a clear definition, research outcomes and their meaning remain muddled and uncertain. As research about a definition is not possible, discussion and reaching consensus is the best way to solve this. A common consensus building method is the Delphi technique.24 The purpose of this technique is to obtain consensus on the opinions of ‘experts’ through question rounds using structured questionnaires. The audience emphasized their preference of working as soon as possible on a definition of MUS.
Workshop participants chose as second most important research question ‘what is the best strategy to recognize MUS in primary care?’ Patients with MUS are functionally impaired and at risk for unnecessary diagnostic procedures and treatments. Recognition of patients with a high risk of persistent MUS is therefore of paramount importance. However, in the absence of a clear definition, studying this research question is difficult.
Thirdly, research about effective interventions in patients with MUS, especially self-management and patient empowerment, should have priority according to 40% of the workshop audience. We should take the cultural background and patient perspectives into account. Probably, more than one intervention is needed to help patients with MUS. Mixed method qualitative research with doctors and patients may be the key to develop an effective intervention for these patients.
Finally, we discussed what doctors in primary care might do wrong, and what they do well, in consultations with patients with MUS. For instance, medical investigations can reassure some patients but can worsen symptoms in other patients. It would be helpful to study predictors in order to decide which strategy will help the individual patient with MUS most.
Themes in MUS research
In Table 3, we grouped the research topic into six themes: (i) definition of MUS, (ii) aetiology and prognosis of MUS, (iii) problems in the consultation with patients with MUS, (iv) therapeutic options for patients with MUS, (v) patient perspectives and (vi) cultural differences. As appears from this table, the most important research themes worth studying were ‘therapeutic options for patients with MUS’ and ‘problems in the consultation with patients with MUS’ (total score 195 and 174, respectively). Research about the definition of MUS got a total score of 120 and research about aetiology and course 115. The research themes ‘cultural differences’ and ‘the patient perspective’ were regarded as less important (total score of 59 and 46, respectively).
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Discussion |
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Bringing together experts of MUS research and discussing the state of the art in MUS research internationally was of great value: we could share the global perspective on MUS and discuss ideas about future developments on MUS research. Although cultural differences will play a role in the classification of patients with MUS, and doctors’ perspectives and management of patients with MUS, we did not find many prominent cultural differences during our symposium and workshop. Doctors from all over the world face more or less the same problems in consultations with patients with MUS. The high attendance rate and the number of nationalities in our symposium and workshop reflect that MUSs are present all over the world and attract great attention of GPs and researchers.
The problem in MUS that was recognized most was the indistinct and ambiguous definition of MUS. There is need for an unambiguous definition of MUS because without a clear definition, research outcomes and their meaning remain muddled and unclear. This need is even more urgent because in the next versions of ICD-11 and DSM-V, the end of the somatoform disorder category is being considered.25,26 Difficulties arising in defining MUS are the connection between MUS and prevalent mental health disorders such as depression and anxiety and functional somatic syndromes such as fibromyalgia and Irritable Bowel Syndrome.27,28 Therefore, integrated research in primary care and specialized services is needed.
Important research themes worth studying appeared to be ‘therapeutic options in patients with MUS’ and ‘problems within the consultation with patients with MUS’. Recent research already focus on these themes.20,29,30 There is need for more patient-oriented research, e.g. qualitative research and mixed method research, to learn more about the needs of patients with MUS.31 Together with studying the course of MUS, this will provide GPs with instructions and advices for the best therapeutic approaches towards these patients.12,13 At best, the results of these studies should be combined with the development and investigation of suitable and effective interventions in these patients.
As far as we know, this is the first time that a research agenda in MUS has been defined by GPs from all over the world. The participation of 29 nationalities from 6 different continents extends the validity of the findings to cultures other than the Western culture. The strength of the nominal group technique as a method for defining the research agenda is its structured, detailed discussion. However, it may also lead to unrepresentative, unreliable judgements and contextual differences, such as differences in national health care systems. Furthermore, the lack of anonymity can make participants feel reserved to articulate their opinion.
We observed an interesting discrepancy: results of research as presented in the symposium focussed on miscommunication between doctors and patients as reason for persisting MUS, whereas in the workshop participants discussed MUS as if it were a real disease with its own aetiology and course. Patients often want to discuss psychosocial problems but doctors do not do so and choose a somatic pathway. This difference between patient's and doctor's perspective is regarded as a cause of persistence of symptoms. Consequently, these symptoms have a ‘course’ and the workshop participants were particularly interested in that course. Only some MUS patients turn out to have a ‘real’ somatic or psychological disease during follow-up. Turning all persistent unexplained symptoms into a disease is medicalizing and not very helpful for patients because they actually want to be heard and discuss their psychosocial problems. If doctors would facilitate this, more MUS might resolve.
Another interesting finding of our workshop was the fact that research on patient views was not considered as priority. Knowledge of patients’ expectations is necessary to answer patients’ needs in consultations of MUS and enhances patients’ satisfaction, adherence and health.32 The discussion on MUS as a real disease and the fact that only doctors participated in defining the research agenda might explain the lack of priority of patient views. Moreover, it shows that patient participation in defining a research agenda is needed in the future.
It is clear from the literature that more research on MUS in primary care is needed in order to provide an improvement of the management of these patients and a better understanding of the problems we face during consultations with patients with MUS. Moreover, it is important to exchange ideas and coordinate research efforts on MUS in primary care at an international level in order to prevent redundancy. Future WONCA conferences, as well as other primary care-oriented conferences, offer excellent opportunities for this purpose.
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Declaration |
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Funding: None.
Ethical approval: None.
Conflicts of interest: None.
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Notes |
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olde Hartman TC, Hassink-Franke LJA, Dowrick C, Fortes C, Lam C, van der Horst HE, Lucassen PLBJ and van Weel-Baumgarten EM. Medically unexplained symptoms in Family Medicine: defining a research agenda. Family Practice 2008; Pages 1–6 of 6.
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