Patients writing about their health condition were abused by a peer reviewer and silenced by The BMJ

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giphyRecently I blogged about authors who were informed by The BMJ that they must keep a review confidential despite having submitted a manuscript under the journal’s laudable policy of open peer review. I did not actually post the review in its entirety because doing so might to make it more difficult for the editors of The BMJ to provide timely, appropriate amends, including an apology to the authors.

I contacted a number of The BMJ editorial staff, starting with the action editor who handled the manuscript. I either got no response or I was told the silencing of the authors concerning their mistreatment was not their doing or anything with which they were willing to intervene.

I am now providing the review, which is patently unprofessional, despite coming from a psychiatrist.

being bmjThe reviewer challenges whether the authors relied on self-diagnosis of chronic fatigue syndrome, perhaps bolstered by doctor shopping until they found agreement.

There were two reviews of this manuscript sent to the authors. Reviewer 1  was succinct and positive:

This is a very well-written and incisive analysis. I would however like to invite authors to comment whether the selection of patients by Oxford criteria might have also partially exaggerated the benefit of trial interventions with graded exercises and CBT.

Should patients submitting manuscripts concerning health conditions provide proof of their diagnoses, such as medical records or letters from their physicians?

Should The BMJ apologize to these patients and their academic collaborator co-authors, given that no such apology has been forthcoming from the Action Editor?

The Editor, Navjoyt Ladher, was trained at Kings College, London where Simon Wessely and Trudie Chalders are faculty.

The reviewer trained at Kings College as well.  He  collaborated  with Simon Wessely on articles concerning chronic fatigue syndrome. I am not revealing his name, but in his review, he indicates that he is a psychiatrist who has mostly practiced in the west of Scotland.

I start by calling attention to some noteworthy points in this 2700 word review. The numbers refer to 13 of the highlighted passages in the review that follows.

  1. The reviewer recommends the manuscript be published without the authors being given the opportunity of revision. The intent of this is that it would draw Rapid Responses protesting what patients with chronic fatigue syndrome have to put up with, including from patients who, like the authors, have the condition.
  2. The reviewer dislikes this paper and yet still wants it to be published.
  3. The reviewer claims the manuscript insults and demeans other patients.
  4. If the paper is published and the PACE investigators don’t respond as the reviewer hopes, the reviewer will post a comment to the authors “Shame on you.”
  5. The authors should just move on and be done with the PACE trial.
  6. The reviewer notes that the paper is billed as a collaboration between patients and scientists, but questions whether any of the authors qualify as “clinicians” or “scientists.”
  7. The reviewer expresses doubts that the patients meet criteria for chronic fatigue syndrome.
  8. The reviewer reiterates the doubt the patients meet criteria for chronic fatigue syndrome and suggests that they were erroneously self-diagnosed.
  9. The reviewer suggests that the authors were erroneously self-diagnosed and went doctor-shopping until they found agreement.
  10. After earlier mentioning that he had not obtained the author’s published review, he questions whether it is a major review.
  11. The reviewer asserts that the PACE investigators can defend the recovery rates they claimed in the PACE trial.
  12. The reviewer questions whether the authors are merely writing about themselves rather than persons with diagnosed chronic fatigue syndrome.
  13. The reviewer claims the authors insult patients with genuine chronic fatigue syndrome when they challenge Wessely’s model emphasizing “fearful cognitions.”

patients should be seenReviewer 2

One of the nice things about providing a referee’s report (and it is even nicer when I am the recipient) is when a fresh look at a manuscript provides simple suggestions that lead to clear improvements in the eventual published paper.  For the 1st time in the three decades that I have been refereeing scientific papers  [1]I am going to recommend publication of a manuscript but only on the condition that no changes are made based on any of my comments or questions or criticisms or praise.  This is because the tone of this paper and certain of its content provide a fascinating illustration of some of the problems that surround the management of people with severe and prolonged fatigue states.  As well as making some quite interesting points these authors provide examples of what people with severe and prolonged fatigue have to put up with – even from other people who themselves have severe and prolonged fatigue.  If this manuscript is published in the BMJ this will give the chance for correspondents in the Rapid Responses section to point out some of its flaws and hopefully a valuable debate will follow that will contribute to a more thoughtful approach to this whole difficult field. 

There has been and continues to be a great deal of scientific controversy surrounding the PACE trial of the treatment of chronic fatigue syndrome(s) using graded exercise therapy vs cognitive behavioural therapy vs adaptive pacing therapy all in addition to standardised care delivered by doctors with specialist experience of chronic fatigue syndrome(s).  The back and forth scientific controversy makes fascinating reading as does this manuscript by …

In my opinion, the differing scientific interpretations of this trial have little or nothing to do with the participants’ scientific training and expertise.  Rather, scientific stances are dependent on people’s personal background and/or their clinical training and/or their clinical experience of assessing and treating patients with severe fatigue states and/or their own personal experience of ill health and/or the illness experience of family members and/or their personal experience of clinical care especially the care they have received from doctors. I will try – briefly and I hope not too boringly – to weave into this referee’s report some of my own background which will perhaps give some understanding of why, overall, [2] I dislike this paper and yet still want it to be published in a widely-read journal.

Whether to publish this paper will not be a straightforward decision for the editors.  It is certainly not an original contribution to the scientific literature.  A paper with similar content by some of the same authors appeared only a couple of months ago in the journal “Fatigue, Biomedicine Health & Behaviour”.  However, if it is published in the BMJ then this would provide a readily accessible update on some of the continuing controversies surrounding the diagnosis, prognosis and treatment of people with severe and prolonged fatigue.  Because of the Rapid Responses/Correspondence section of the BMJ it will give the investigators in the PACE trial the opportunity to put their different scientific interpretations to a wider audience.  They have already published these different interpretations in “Fatigue, Biomedicine, Health & Behaviour”.  I am still waiting for that paper from my librarian but I am certain that these scientists are more than able to defend their trial.  An up to date defence of the PACE trial with balanced consideration of its strengths and weaknesses will be very helpful for clinicians and policy makers if it is published in the readily accessible BMJ.com.  This will, in turn, make it easier when clinicians and health service managers try to improve services for the wide range of patients with severe and prolonged fatigue.

The main reason I would like this paper to be accepted is because of some careless use of language by the authors.  This can happen to anybody and I am sure these authors do not really mean what they say.  [3]They have however insulted and demeaned a subgroup of people with severe and prolonged fatigue (see below).  This is in spite of the fact that [the authors] are themselves in poor health due to severely fatiguing illnesses.  [4] I am sure this will be picked up by the PACE investigators and other readers of the BMJ and highlighted in the Rapid Responses.  If not, then I will write in and tell the authors that they should know better and I think I would end with a “Shame on you”.   

If this manuscript is published in the BMJ and the authors receive the appropriate criticism (and credit) for some the things they have said I hope they will then get on with applying their skills and intelligence and insights to some proper work that will have a chance of genuinely benefiting patients with chronic fatigue syndromes and other overlapping disorders.  [5]It is about time that they moved on from their obsessive (in the non-psychiatric use of the term) poring over the results of a good (albeit imperfect) randomised controlled trial (…and name me one perfect trial that exists in any clinical field).

Here are some comments on the manuscript that may be helpful to the editors in deciding whether or not to publish this non-original paper in a major general journal.  I am hoping that these comments will help draw out how interesting this paper will be to many readers of the BMJ.

1)            These authors make no attempt whatsoever to acknowledge the heterogeneity of patients who are labelled with a diagnosis of chronic fatigue syndrome (and, in my experience, the even greater heterogeneity of the smaller number of patients who are labelled using the diagnostic concept of myalgic encephalomyelitis).

2)           [6] These authors make a big deal of the fact that their paper is the result of collaboration between patients and scientists.  I am still unsure whether that should be clinical scientists.  [authors]– do you have any clinical training and experience?

3)            From what I can gather from this paper and their other writings [authors]  seem to believe that the best definition of chronic fatigue syndrome is whatever condition it is that has led to ill health in [the authors].

4)           [7]  I am sorry to hear of these authors’ ill health.  I hope they will not be upset when I say that I do not accept their diagnosis of chronic fatigue syndrome.  I do not accept at face value anybody’s declared diagnosis of chronic fatigue syndrome until I have done my own history and examination.  The reasons for this are as follows.  When I was a junior registrar in neurology in 1981/1982 our team investigated referrals – including self referrals – of patients with severe fatigue.  They received a battery of investigations from brain scans through lumbar puncture through visual evoked responses to muscle biopsy.  Every test would be normal.  We never took a social history and never carried out a mental state examination.  Come to think of it we never even took a proper past medical history.  The patients would then be told that they had a condition called myalgic encephalomyelitis and would be sent home to rest with the prognosis that they would not improve but that there may be a cure in the future with advances in neurovirology.  As I have written before .. and I have thought to myself on many occasions since – may God forgive me for the part I played in destroying the lives of some of these vulnerable patients. 

I later did clinical work with referrals with severe fatigue in three different clinical and geographical settings between 1988 and 2011.  I found then that a substantial minority (for a period it was the majority) of patients with a diagnosis of chronic fatigue syndrome and myalgic encephalomyelitis had readily diagnosable conditions using basic knowledge of general medicine and general psychiatry.  Sometimes my rediagnosis/reformulation would lead on to effective treatment.  However, some patients would reject these alternative explanations.  Some, sadly, would be angry towards me and state that I was not taking their illness seriously.  It was very disconcerting and it raised major worries about the inadequacies of my clinical communication skills when I would tell a patient with a diagnosis of chronic fatigue syndrome or, more usually, myalgic encephalomyelitis that their profound fatigue and other serious symptoms were better explained by, for example, their recurrent diabetic ketoacidosis; or the systemic effects of their known severe rheumatoid arthritis and its treatment; or their severe psychotic illness and its treatment; or profound depressive illness; or crippling panic disorder; or Reiter’s disease; or obsessive compulsive disorder with co-morbid depression; or malnutrition due to anorexia nervosa; or the temporary aftermath of newly diagnosed and treated severe thyroid disease…….I could go on and on – and then be told by the patient that I was not taking their ill health seriously.

I accept that my experience must have been unrepresentative since I was working from psychiatric outpatient clinics and for most of this time I was based in the West of Scotland that was the epicentre of myalgic encephalomyelitis movement.  However, I cannot believe that I am the only doctor who encountered this phenomenon.  How can I be sure that [the authors]  do not have ill health that could be much better categorised using any one of a range of much more straightforward diagnoses?

5)            [8] Who diagnosed [the authors’] condition?  I do not expect an answer since their medical history is and should remain confidential.  However I have to raise the possibility that they are self diagnosed.  For a while at my clinic a clear majority of diagnoses of chronic fatigue syndrome or myalgic encephalomyelitis had been made by the patient themselves or by a relative or friend or neighbour and not by any doctor or other clinician.  Many (but not all) of these patients had other obvious reasons for their fatigue after taking a full history and doing a physical examination and mental state examination.  I accept that such high frequency of self diagnosis/lay diagnosis may not be found in other clinics but, once again, I cannot believe that I am the only clinician who has encountered this phenomenon.

6)           [9]If they have been diagnosed with chronic fatigue syndrome by a doctor was this by their own doctor?  There certainly used to be a phenomenon whereby patients with a self diagnosis of chronic fatigue syndrome and (even more so) myalgic encephalomyelitis used to go doctor shopping until they found a doctor who agreed with their own diagnosis.  In my experience many (but not all) of these patients had readily diagnosable and fairly straightforward alternative diagnoses. 

7)            I notice in the manuscript that [one of the authors]  says she has “recently co-authored a major critical review of the concept of psychological causation in medicine…”.[10] [Author], I mean no disrespect, but is it for you to say that your review is a major one?  I hope you will not mind my saying that I found it to be very unbalanced and highly selective in its use of the medical literature.  Once again, I want this manuscript as it stands to appear in a widely-read journal so that interested readers will be able to get a clearer view of [this author’s]  thinking and I hope it will be criticised appropriately.

8)            I think [the authors]  are right to question the number of subjects in whom there has been “recovery”.  They may not be aware that the word “recovery” has been hijacked throughout mental health services by the very influential and international Recovery Movement.  This is a positive example of a powerful and creative collaboration between service users and clinicians.  [The authors]  should look it up.  The Recovery Movement has contributed to improved care for many patients especially those with chronic psychotic illnesses.  I think I was the last person involved in mental health services in Scotland – patient or clinician – to argue against some uncritical approaches of the Recovery Movement but I ended up giving in.  I find myself talking to certain patients about recovery – who have no chance of recovery in the dictionary sense of the word.  [11]The PACE investigators will easily be able to defend themselves in regard to recovery in their trial since they have operationally defined what they meant by their use of this word and they have been very clear about changes they made in their definitions.  Nevertheless, [An author] and colleagues do have a point about how reported “recovery rates” could be misleading.     

9)    [The authors] are right that the PACE trial is not definitive.  I have only seen the PACE investigators say this on one occasion and in one paper although I may have missed some other instances.  I think this was just some careless use of language.  Give them a break.

10)   There is something unpleasant in the tone of this article (although I am also being influenced here by outpourings on the Internet).  I cannot help but get the impression that the authors were punching the air when they thought they had come up with support for their views that certain treatment methods for certain people with serious ill health were not as helpful as others (both patients and clinicians) had hoped.  [12] Are [the patient authors]  absolutely sure that they are writing about syndromes of chronic fatigue?  Are they sure they are not simply writing about themselves?

11)         [13] This brings me to the insulting language about certain patients with severe and prolonged fatigue states.  [The patient authors]  are absolutely certain that their condition is not influenced by psychological troubles or social stressors.  This does not give them and their co-authors any right to belittle other patients and understate the severity of illness in those with a diagnosis of chronic fatigue syndrome where such factors are playing a major part in their ill health.  These authors write “…the CBT programme considers patients’ concerns about exercise to be merely ‘fearful cognitions’ that need addressing”.  Earlier in the article they state “This model proposes that there is no major ongoing disease process in CFS – merely deconditioning due to recent inactivity, and its various consequences”.  Merely?  Have they never met anybody with near 100% disability due to mere fearful cognitions?  I have.  Have they never spoken with patients who are at risk of dying due to mere fearful cognitions and their consequences?  I have.  XWilshire and her colleagues should be ashamed of themselves and I think their shameful language should be published and then condemned.  They should then be given the opportunity to write what they really mean and apologise to those patients about whom they have been so offensive.  This is the main reason why I want this article to appear in a widely read general journal that has an active Correspondence/Rapid Responses section. 

I hope these comments are helpful to the editors.  There is such widespread and heated debate around this subject that I think it would be helpful for some of it to appear in a widely-read journal.  I hope the editors will agree with me and publish this article – then stand back and see how the debate unfolds in the Rapid Responses/Correspondence.  I think this will lead in the end to more people with severe and chronic fatigue getting better care packages – which will include for some cognitive behavioural therapy and for others graded exertion therapy and for yet others perhaps even adaptive pacing therapy and for others none of the above but with high quality informed medical care for everybody.  Hopefully it will also contribute to efforts to raise funding for more research projects that will join the PACE trial in giving us guidance about how to treat and how not to treat individual patients with severe fatigue be it explained or unexplained or simple or complicated.