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Does myalgic encephalomyelitis exist?
CORRESPONDENCE
Does myalgic encephalomyelitis exist? Sir—Olivier Mouterde (Feb 17, p 562)1 states that he has never encountered myalgic encaphalomyelitis (ME) in children in France, Belgium, or Switzerland, whereas it is common in the UK. He suggests genetic differences as an explanation, but, in all likelihood, the recognition of the disorder is culturally influenced. Hacking’s work2 has helped us to understand illnesses that find homes in a given place and time. He proposed the idea of an ecological niche, the setting in which a mental illness thrives. For example, in France in the 1880s mad travellers, or fuguers, flourished. These compulsive travellers were, however, confined to France and other countries on the European continent and not in the UK and USA. Many reasons have been suggested, including the lack of conscript armies in the UK and USA, so that no escape was needed; furthermore, fuguers were seen as degenerate in France, but not elsewhere. By 1910, the disorder had died out. What ecological factors have allowed ME to flourish in the UK in children and adults? The behaviour of doctors and the media have affected the ways in which illnesses become manifest. Shorter’s account3 of psychologically induced illness shows how patients throughout the 20th century reported physical symptoms corresponding to the current models of disease espoused by psychiatrists. The most striking example of this was the demise of the florid Charcot-style hysteria after Babinski’s attack in 1908. Most doctors are surprisingly unaware of their potential to influence the course of an individual patient’s illness. In one study, of adult patients with severe non-organic disability (some with chronic fatigue syndrome [CFS]), four patients who believed (erroneously) that multiple sclerosis accounted for their symptoms also had difficulties with bladder control, requiring catheterisation.4 Specific and direct questions about urinary function could have affected the development and appearance of the symptoms, presumably because of a suggestion and fear in people willing to embrace the sick role. Mouterde also makes the valid point that most children have heard about ME before developing it and that they know they can catch it
THE LANCET • Vol 357 • June 9, 2001
because they can read about it. The media undoubtedly has a role in publicising this illness in children, but what the media cannot explain is the continued prevalence of motor conversion symptoms (hysterical paralyses and sensory abnormalities) in adults. 3 years ago Hacking stated that hysteria does not exist as a possible diagnosis today, but evidence strongly refutes this view.5 Most neurologists see at least one patient a month with conversion hysteria in the outpatient clinic, and continue to do so in the 21st century. How do the medical historians explain the continued existence of this illness, which is invariably concealed from the patient and receives no exposure in the media? Christopher Bass Department of Psychological Medicine, John Radcliffe Hospital, Oxford OX3 9DU, UK 1 2 3
4
5
Mouterde O. Myalgic encephalomyelitis in children. Lancet 2001; 357: 562. Hacking I. Mad Travellers. London: Free Association Books, 1998. Shorter E. From paralysis to fatigue: a history of psychosomatic illness in the modern era. New York: Free Press, 1992. Davison P, Sharpe M, Wade D, Briss C. Wheelchair patients with non-organic disease: a psychological enquiry. J Psychosom Res 1999; 47: 93–103. Akagi H, House A. The epidemiology of hysterical conversion. In: Halligan P, Bass C, Marshall J, eds. Contemporary approaches to the study of hysteria. Oxford: Oxford Univerisity Press 2001: 73–87.
Sir—As a Belgian liaison-psychiatrist, who has seen more than 2000 patients with chronic, non-organically explained fatigue and pain, I have ambivalent feelings about the Olivier Mouterde’s criticism (Feb 17, p 562)1 of the diagnosis of myalgic encephalomyelitis (ME) in children. He is right to call attention (as others have done2) to the sociocultural determination of illness, including the impact of illness labels on the illness itself and health-care-seeking behaviour. There is a substantial difference in my country between the Dutch-speaking and the Frenchspeaking populations. French-speaking Belgian colleagues have repeatedly confirmed that the chronic fatigue syndrome (CFS) has not yet become a hyped illness, as it is in Flanders (as well as in the Netherlands and the UK). In Flanders, for example, there are currently four CFS/ME self-help groups, whereas none exists in Wallonia. The Flemish press, has regularly reported the disorder for 10 years (eg, describing patients’ experiences about disbelief and
incomprehension, or about so-called research breakthroughs), but the Walloon press has shown interest only in the past few years. Walloons are more oriented— culturally as well as scientifically—to France, whereas Flemish people are on the same wavelength as countries with Anglo-Saxon culture and science. Consequently, Walloon and Flemish patients and doctors use different labels to communicate about distress and ill health. This labelling process is strongly mediated by the media but, interestingly, a new phenomenon is that the patients and their self-help associations actively participate in this process I disagree, however, with Mouterde’s suggestion that CFS is not a serious health issue in adults as well as in adolescents and children. In more-developed countries, stressrelated illnesses and associated healthcare-seeking behaviour seems to have increased. Most likely, the innovative and bridging sciences of psychoneuroendocrinology and immunology will improve our understanding of the neurobiological substrate of all forms of ill health, and lay the basis for effective treatment.3 However, the fact is not altered that it would be interesting to investigate this disease from a broader medical, social, and sociocultural perspective. Meanwhile, labels such as CFS and ME should be used in a constructive way, not as a condemnation of disability, but as a point of departure for realistic and pragmatic help.4 Boudewijn van Houdenhove Department of Psychosomatic Rehabilitation, University Hospitals, KU Leuven, B-3212 Lubbeek, Belgium 1 2
3 4
Mouterde O. Myalgic encephalomyelitis in children. Lancet 2001; 357: 562. Ware NC, Kleinman A. Culture and somatic experience: the social course of illness in neurasthenia and the chronic fatigue syndrome. Psychosom Med 1992; 54: 546–60. Van Houdenhove B. Psychosocial stress and pain. Eur J Pain 2001; 4: 225–28. Van Houdenhove B. Moe in tijden van stress: luisteren naar het chronischevermoeidheidssyndroom. [Tired in times of stress: listening to the chronic fatigue syndrome]. Tielt: Lannoo, 2001.
Sir—Olivier Mouterde1 asks, tongue in cheek, whether ME might be due to a virus confined in some mysterious way by the English Channel, or perhaps whether it results from a genetic peculiarity of UK children. He speculates that it may be a culturally induced disorder of some sort, with no organic basis. Many other observers of ME have wondered about its cause, but its
1889
For personal use. Only reproduce with permission from The Lancet Publishing Group.
CORRESPONDENCE
geographical dispersion doubtless extends further than the UK. Since the descriptions of 198 cases among the staff of Los Angeles County General Hospital, USA, in 19342 and the outbreak of more than 400 cases in the town of Akureyri, Iceland, in 1948–493 there have been reports of sporadic and epidemic occurrences of similar illnesses in the USA, the UK, Germany, Greece, South Africa, and Australia.4 The terms Akureyri disease, Akureyri fever, Icelandic disease, and benign myalgic encephalitis have all been used, and these seem to be the same disorder now termed chronic fatigue syndrome. Hyde and Bergmann5 provided a useful 40-year follow-up to the original paper from Iceland. Mouterde reminds us that the loss of long-term memory can be as much of an issue in a profession as it is in a patient. Elmer R Grossman 899 Euclid Avenue, Berkeley, CA 94708, USA 1 2
3
4
5
Mouterde O. Myalgic encaphalomyelitis in children. Lancet 2001; 357: 562. Gilliam AG. Epidemiological study of an epidemic, diagnosed as poliomyelitis, occurring among the personnel of Los Angeles County General Hospital during the summer of 1934. US Public Health Bull 1938; 240: 1–90. Sigurdsson B, Sigurjónsson J, Sigurdsson JHJ, Thorkelsson J, Gudmundsson KR. A disease epidemic in Iceland simulating poliomyelitis. Am J Hyg 1950; 52: 222–38. McEvedy CP, Beard AW. Concept of benign myalgic encephalitis. BMJ 1970; 1: 11–15. Hyde B, Bergmann S. Akureyri disease (myalgic encephalomyelitis), forty years later. Lancet 1988; 2: 1191.
Sir—Olivier Mouterde1 has clearly been impressed by the genuineness and severity of some cases of ME in the UK, and yet has a clinical impression that the disorder does not exist in France, Belgium, or Switzerland. We think that Mouterde’s observations are related to differences in diagnostic labelling by doctors in those countries. We are paediatricians in the UK and have been asked to give many second opinions on cases. If families suggest the diagnosis to their doctors, they are told that ME does not exist. When investigations come back negative, parents are told that the child’s disorder must be psychological and are referred to child psychiatry. Doctors seem to believe it is not medically respectable to diagnose ME. Indeed, the report of the Royal Colleges (to which Mouterde refers) expressly attempted to abolish the
1890
term ME and replace it with the far weaker euphemism of CFS. The controversy surrounding terminology remains unresolved. We prefer the term ME, since it retains the power of the original name. Paediatric textbooks in the UK hardly ever include ME in their index, although most make some mention of CFS. One major reference text2 includes CFS only in the chapter on child psychiatry. Despite these continuing controversies, many family doctors and paediatricians in this country are happy and confident to make a positive clinical diagnosis of ME or CFS. There are strong patient support organisations. There is a Working Party on ME and CFS set up by our previous Sir Kenneth Calman that is due to report shortly, and the Royal College of Paediatrics and Child Health is doing epidemiological research on the incidence in children. A further factor handicapping the diagnosis of ME is specialisation of paediatricians. Because ME can present with such multituple symptoms, children are frequently referred to subspecialists for their most prominent symptoms. These specialists do a battery of normal investigations, but miss the diagnosis and discharge the patients back to the family doctor. Another factor is lack of time. The diagnosis of ME is essentially based on history, there being no consistent physical signs or abnormal tests. Because the child may have up to 20 presenting symptoms, it can take longer than 1 h to take the history. The temptation to take short cuts and proceed to investigations can be irresistible. We met a distinguished French gastroenterologist who was due to give a talk on recurrent abdominal pain. We asked his views on abdominal migraine, which made him look nervous. He lowered his voice and said this was not a respectable diagnosis in France and people get laughed at if they use the term. This attitude seems to parallel that for ME. We strongly suspect that Mouterde will find himself making the diagnosis in French children in the not too distant future. *Nigel Speight, Alan Franklin Department of Paediatrics, Dryburn Hospital, Durham DH1 5TW, UK 1 2
Mouterde O. Myolgic encepholomyelitis in children. Lancet 2001: 357: 562. Forfar and Arneil’s Textbook of Paediatrics, 5th edn. London: Churchill Livingstone, 1998.
ABC of secondary prevention Sir—I was intrigued by the use of the ABCDE of secondary prevention by Jerome Cohen1 in his March 31 commentary. For the past few years I have been encouraging my house staff to think of the “A to H” of secondary prevention after myocardial infarction, and have been gratified by the obvious value to them. Evidence includes mutterings of “what was C again?” at the end of the bed during ward rounds. My extensions to Cohen’s suggestion are: A aspirin. B -blockers. C cholesterol and cigarettes. D dysfunction (left ventricular—so the issue of ACE inhibitors is clearly identified in the postmyocardialinfarction population). E education and exercise (test). F follow-up (added in to get the house staff to . . .). G . . . GTN (glyceryl trinitrate), to be added to the discharge prescription. H home, just to round it off nicely. In a system where the house staff come and go very quickly (Coronary Care Unit house staff are with us for 1 week), I believe this type of approach can help even excellent doctors find their way more effectively. Philip C Adams Department of Cardiology, Ward 50 Office, Royal Victoria Infirmary, Newcastle upon Tyne NE1 4LP, UK 1
Cohen JD. ABCs of secondary prevention of CHD: easier said than done. Lancet 2001; 357: 972–73.
DEPARTMENT OF ERROR Persistence of sulphonamide resistance in Escherichia coli in the UK despite national prescribing restriction—In this Article by Virve I Enne and colleagues (April 28, p 1325), the numbers of sulphonamide prescriptions given in the Findings section of the Summary and in the first paragraph of the Results section (p 1326), should have been 3 208 000 in 1991 and 77 000 in 1999. Effect of fenofibrate on progression of coronaryartery disease in type 2 diabetes: the Diabetes Atherosclerosis Intervention Study, a randomised study—In this Article by the Diabetes Atherosclerosis Intervention Study Investigators (Mar 24, p 905), figure 2 on page 907 should have said that 19 patients in the fenofibrate group did not undergo the final angiogram and had data imputed.
THE LANCET • Vol 357 • June 9, 2001
For personal use. Only reproduce with permission from The Lancet Publishing Group.
Does myalgic encephalomyelitis exist? Sir—Olivier Mouterde (Feb 17, p 562)1 states that he has never encountered myalgic encaphalomyelitis (ME) in children in France, Belgium, or Switzerland, whereas it is common in the UK. He suggests genetic differences as an explanation, but, in all likelihood, the recognition of the disorder is culturally influenced. Hacking’s work2 has helped us to understand illnesses that find homes in a given place and time. He proposed the idea of an ecological niche, the setting in which a mental illness thrives. For example, in France in the 1880s mad travellers, or fuguers, flourished. These compulsive travellers were, however, confined to France and other countries on the European continent and not in the UK and USA. Many reasons have been suggested, including the lack of conscript armies in the UK and USA, so that no escape was needed; furthermore, fuguers were seen as degenerate in France, but not elsewhere. By 1910, the disorder had died out. What ecological factors have allowed ME to flourish in the UK in children and adults? The behaviour of doctors and the media have affected the ways in which illnesses become manifest. Shorter’s account3 of psychologically induced illness shows how patients throughout the 20th century reported physical symptoms corresponding to the current models of disease espoused by psychiatrists. The most striking example of this was the demise of the florid Charcot-style hysteria after Babinski’s attack in 1908. Most doctors are surprisingly unaware of their potential to influence the course of an individual patient’s illness. In one study, of adult patients with severe non-organic disability (some with chronic fatigue syndrome [CFS]), four patients who believed (erroneously) that multiple sclerosis accounted for their symptoms also had difficulties with bladder control, requiring catheterisation.4 Specific and direct questions about urinary function could have affected the development and appearance of the symptoms, presumably because of a suggestion and fear in people willing to embrace the sick role. Mouterde also makes the valid point that most children have heard about ME before developing it and that they know they can catch it
THE LANCET • Vol 357 • June 9, 2001
because they can read about it. The media undoubtedly has a role in publicising this illness in children, but what the media cannot explain is the continued prevalence of motor conversion symptoms (hysterical paralyses and sensory abnormalities) in adults. 3 years ago Hacking stated that hysteria does not exist as a possible diagnosis today, but evidence strongly refutes this view.5 Most neurologists see at least one patient a month with conversion hysteria in the outpatient clinic, and continue to do so in the 21st century. How do the medical historians explain the continued existence of this illness, which is invariably concealed from the patient and receives no exposure in the media? Christopher Bass Department of Psychological Medicine, John Radcliffe Hospital, Oxford OX3 9DU, UK 1 2 3
4
5
Mouterde O. Myalgic encephalomyelitis in children. Lancet 2001; 357: 562. Hacking I. Mad Travellers. London: Free Association Books, 1998. Shorter E. From paralysis to fatigue: a history of psychosomatic illness in the modern era. New York: Free Press, 1992. Davison P, Sharpe M, Wade D, Briss C. Wheelchair patients with non-organic disease: a psychological enquiry. J Psychosom Res 1999; 47: 93–103. Akagi H, House A. The epidemiology of hysterical conversion. In: Halligan P, Bass C, Marshall J, eds. Contemporary approaches to the study of hysteria. Oxford: Oxford Univerisity Press 2001: 73–87.
Sir—As a Belgian liaison-psychiatrist, who has seen more than 2000 patients with chronic, non-organically explained fatigue and pain, I have ambivalent feelings about the Olivier Mouterde’s criticism (Feb 17, p 562)1 of the diagnosis of myalgic encephalomyelitis (ME) in children. He is right to call attention (as others have done2) to the sociocultural determination of illness, including the impact of illness labels on the illness itself and health-care-seeking behaviour. There is a substantial difference in my country between the Dutch-speaking and the Frenchspeaking populations. French-speaking Belgian colleagues have repeatedly confirmed that the chronic fatigue syndrome (CFS) has not yet become a hyped illness, as it is in Flanders (as well as in the Netherlands and the UK). In Flanders, for example, there are currently four CFS/ME self-help groups, whereas none exists in Wallonia. The Flemish press, has regularly reported the disorder for 10 years (eg, describing patients’ experiences about disbelief and
incomprehension, or about so-called research breakthroughs), but the Walloon press has shown interest only in the past few years. Walloons are more oriented— culturally as well as scientifically—to France, whereas Flemish people are on the same wavelength as countries with Anglo-Saxon culture and science. Consequently, Walloon and Flemish patients and doctors use different labels to communicate about distress and ill health. This labelling process is strongly mediated by the media but, interestingly, a new phenomenon is that the patients and their self-help associations actively participate in this process I disagree, however, with Mouterde’s suggestion that CFS is not a serious health issue in adults as well as in adolescents and children. In more-developed countries, stressrelated illnesses and associated healthcare-seeking behaviour seems to have increased. Most likely, the innovative and bridging sciences of psychoneuroendocrinology and immunology will improve our understanding of the neurobiological substrate of all forms of ill health, and lay the basis for effective treatment.3 However, the fact is not altered that it would be interesting to investigate this disease from a broader medical, social, and sociocultural perspective. Meanwhile, labels such as CFS and ME should be used in a constructive way, not as a condemnation of disability, but as a point of departure for realistic and pragmatic help.4 Boudewijn van Houdenhove Department of Psychosomatic Rehabilitation, University Hospitals, KU Leuven, B-3212 Lubbeek, Belgium 1 2
3 4
Mouterde O. Myalgic encephalomyelitis in children. Lancet 2001; 357: 562. Ware NC, Kleinman A. Culture and somatic experience: the social course of illness in neurasthenia and the chronic fatigue syndrome. Psychosom Med 1992; 54: 546–60. Van Houdenhove B. Psychosocial stress and pain. Eur J Pain 2001; 4: 225–28. Van Houdenhove B. Moe in tijden van stress: luisteren naar het chronischevermoeidheidssyndroom. [Tired in times of stress: listening to the chronic fatigue syndrome]. Tielt: Lannoo, 2001.
Sir—Olivier Mouterde1 asks, tongue in cheek, whether ME might be due to a virus confined in some mysterious way by the English Channel, or perhaps whether it results from a genetic peculiarity of UK children. He speculates that it may be a culturally induced disorder of some sort, with no organic basis. Many other observers of ME have wondered about its cause, but its
1889
For personal use. Only reproduce with permission from The Lancet Publishing Group.
CORRESPONDENCE
geographical dispersion doubtless extends further than the UK. Since the descriptions of 198 cases among the staff of Los Angeles County General Hospital, USA, in 19342 and the outbreak of more than 400 cases in the town of Akureyri, Iceland, in 1948–493 there have been reports of sporadic and epidemic occurrences of similar illnesses in the USA, the UK, Germany, Greece, South Africa, and Australia.4 The terms Akureyri disease, Akureyri fever, Icelandic disease, and benign myalgic encephalitis have all been used, and these seem to be the same disorder now termed chronic fatigue syndrome. Hyde and Bergmann5 provided a useful 40-year follow-up to the original paper from Iceland. Mouterde reminds us that the loss of long-term memory can be as much of an issue in a profession as it is in a patient. Elmer R Grossman 899 Euclid Avenue, Berkeley, CA 94708, USA 1 2
3
4
5
Mouterde O. Myalgic encaphalomyelitis in children. Lancet 2001; 357: 562. Gilliam AG. Epidemiological study of an epidemic, diagnosed as poliomyelitis, occurring among the personnel of Los Angeles County General Hospital during the summer of 1934. US Public Health Bull 1938; 240: 1–90. Sigurdsson B, Sigurjónsson J, Sigurdsson JHJ, Thorkelsson J, Gudmundsson KR. A disease epidemic in Iceland simulating poliomyelitis. Am J Hyg 1950; 52: 222–38. McEvedy CP, Beard AW. Concept of benign myalgic encephalitis. BMJ 1970; 1: 11–15. Hyde B, Bergmann S. Akureyri disease (myalgic encephalomyelitis), forty years later. Lancet 1988; 2: 1191.
Sir—Olivier Mouterde1 has clearly been impressed by the genuineness and severity of some cases of ME in the UK, and yet has a clinical impression that the disorder does not exist in France, Belgium, or Switzerland. We think that Mouterde’s observations are related to differences in diagnostic labelling by doctors in those countries. We are paediatricians in the UK and have been asked to give many second opinions on cases. If families suggest the diagnosis to their doctors, they are told that ME does not exist. When investigations come back negative, parents are told that the child’s disorder must be psychological and are referred to child psychiatry. Doctors seem to believe it is not medically respectable to diagnose ME. Indeed, the report of the Royal Colleges (to which Mouterde refers) expressly attempted to abolish the
1890
term ME and replace it with the far weaker euphemism of CFS. The controversy surrounding terminology remains unresolved. We prefer the term ME, since it retains the power of the original name. Paediatric textbooks in the UK hardly ever include ME in their index, although most make some mention of CFS. One major reference text2 includes CFS only in the chapter on child psychiatry. Despite these continuing controversies, many family doctors and paediatricians in this country are happy and confident to make a positive clinical diagnosis of ME or CFS. There are strong patient support organisations. There is a Working Party on ME and CFS set up by our previous Sir Kenneth Calman that is due to report shortly, and the Royal College of Paediatrics and Child Health is doing epidemiological research on the incidence in children. A further factor handicapping the diagnosis of ME is specialisation of paediatricians. Because ME can present with such multituple symptoms, children are frequently referred to subspecialists for their most prominent symptoms. These specialists do a battery of normal investigations, but miss the diagnosis and discharge the patients back to the family doctor. Another factor is lack of time. The diagnosis of ME is essentially based on history, there being no consistent physical signs or abnormal tests. Because the child may have up to 20 presenting symptoms, it can take longer than 1 h to take the history. The temptation to take short cuts and proceed to investigations can be irresistible. We met a distinguished French gastroenterologist who was due to give a talk on recurrent abdominal pain. We asked his views on abdominal migraine, which made him look nervous. He lowered his voice and said this was not a respectable diagnosis in France and people get laughed at if they use the term. This attitude seems to parallel that for ME. We strongly suspect that Mouterde will find himself making the diagnosis in French children in the not too distant future. *Nigel Speight, Alan Franklin Department of Paediatrics, Dryburn Hospital, Durham DH1 5TW, UK 1 2
Mouterde O. Myolgic encepholomyelitis in children. Lancet 2001: 357: 562. Forfar and Arneil’s Textbook of Paediatrics, 5th edn. London: Churchill Livingstone, 1998.
ABC of secondary prevention Sir—I was intrigued by the use of the ABCDE of secondary prevention by Jerome Cohen1 in his March 31 commentary. For the past few years I have been encouraging my house staff to think of the “A to H” of secondary prevention after myocardial infarction, and have been gratified by the obvious value to them. Evidence includes mutterings of “what was C again?” at the end of the bed during ward rounds. My extensions to Cohen’s suggestion are: A aspirin. B -blockers. C cholesterol and cigarettes. D dysfunction (left ventricular—so the issue of ACE inhibitors is clearly identified in the postmyocardialinfarction population). E education and exercise (test). F follow-up (added in to get the house staff to . . .). G . . . GTN (glyceryl trinitrate), to be added to the discharge prescription. H home, just to round it off nicely. In a system where the house staff come and go very quickly (Coronary Care Unit house staff are with us for 1 week), I believe this type of approach can help even excellent doctors find their way more effectively. Philip C Adams Department of Cardiology, Ward 50 Office, Royal Victoria Infirmary, Newcastle upon Tyne NE1 4LP, UK 1
Cohen JD. ABCs of secondary prevention of CHD: easier said than done. Lancet 2001; 357: 972–73.
DEPARTMENT OF ERROR Persistence of sulphonamide resistance in Escherichia coli in the UK despite national prescribing restriction—In this Article by Virve I Enne and colleagues (April 28, p 1325), the numbers of sulphonamide prescriptions given in the Findings section of the Summary and in the first paragraph of the Results section (p 1326), should have been 3 208 000 in 1991 and 77 000 in 1999. Effect of fenofibrate on progression of coronaryartery disease in type 2 diabetes: the Diabetes Atherosclerosis Intervention Study, a randomised study—In this Article by the Diabetes Atherosclerosis Intervention Study Investigators (Mar 24, p 905), figure 2 on page 907 should have said that 19 patients in the fenofibrate group did not undergo the final angiogram and had data imputed.
THE LANCET • Vol 357 • June 9, 2001
For personal use. Only reproduce with permission from The Lancet Publishing Group.