A preschool boy had a biopsy to confirm a condition from which he subsequently made a complete recovery. Later, he and his younger brother were reported by their female carer to have developed a possible recurrence and this looked likely on near-patient testing. At each review their carer urged us to undertake further biopsies on them both, but we did not feel that this was necessary.
Their carer also reported that they were receiving treatment for three other disparate conditions and one suffered from a fourth unpleasant symptom.
During a hospital admission the near-patient test we were performing repeatedly was always abnormal when his carer was on the ward, and normal when she was not. Further testing of the samples in question showed contamination with female genetic material. The carer admitted to deliberately contaminating the samples to achieve a positive test.
While this condition is well described, it has not been identified by karyotyping in this way before. This probably accounted for us being slow to diagnose it. There are extremely important reasons for diagnosing fabricated illnesses.
Unsurprisingly, the carer was not prepared to give consent for publication. Despite anonymising the case, editors of medical journals are unprepared to publish it without her consent. As a result of this decision, this new diagnostic information will not be disseminated to other paediatricians, which may contribute to other children being unnecessarily exposed to undiagnosed abuse. There will be many other similar examples.
The Forum agreed that in cases such as this, many editors of medical journals in the UK are unlikely to publish the study without consent. Appealing to editors to publish the case report in its current form on the grounds that it is in the public interest is not likely to be successful as it might prove difficult to balance the benefit to society against the rights of the family. Three suggestions were put forward. (1) The paper could be submitted to a journal outside of the UK. (2) The editor could publish an anonymous annotation on the subject and hence this new diagnostic information would be disseminated to other paediatricians. (3) The laboratory test used, without any patient details, could be reported—perhaps as a letter—again allowing dissemination of the information without revealing the specific case.
A UK journal has now agreed to publish a letter from us that outlines the medical issues involved in this case and discusses the potential problems in future publications because of the legal position relating to case reports.
We received a paper reporting on the outcomes of treatment of an individual with obsessive–compulsive disorder and body dysmorphic disorder, which seemed not to respond to standard treatment. Following ethical review and approval, and individual consent, the individual was treated with several distinct courses of an experimental therapy. The individual’s clinical and family history, and their outcomes in response to the experimental therapy, were detailed in the paper. The editors felt that sufficient details were included to potentially identify the patient, so as outlined in our editorial policies, we asked the authors to obtain signed consent to publication from the patient.
The authors responded that they had attempted to remove all identifiers from the paper (although the editors felt that very substantial and identifying details remained), and that revealing the patient’s identity to the editorial office would violate their clinical protocol, and the principle of patient anonymity. The editors discussed this and felt that it was still very important that the patient have read the paper and agreed to its publication; secondly however, we were concerned about the journal having access to (and our ability to keep confidential) the patients’ names. We also wondered whether under HIPAA law it would even be legal for the authors to give us the patient’s identity.
Therefore, the editors proposed that we would ask the author to have the patient (or patient’s parents) sign our consent-to-publication form, and then include in the paper a statement that they had done so, but that we stop asking authors to send us copies of these forms and simply rely on the authors’ word instead. Is this acceptable?
The Forum agreed that this was a curious case and raised issues relating to HIPAA law. Some argued that there is no need for a journal to know the identity of the patients reported in case reports. The authors could be asked for a statement that they have followed the correct procedures and obtained consent from the patient. But there is no need for the journal to see the consent form and hence no need to reveal the identity of the patient. If a problem arises in the future, the journal would have a reasonable defence as the onus would be on the authors to have obtained consent. The editor could request a signed assertion from the authors that they have obtained consent. Although the journal’s own consent form can be used, the Forum noted that it would be very useful to have a generic consent form so that patients need only sign a form once in the event that the paper is rejected by one journal and submitted to another journal. The Forum agreed that the editor’s proposed course of action is acceptable.
The authors were asked to confirm in their paper that the patient had seen the manuscript and agreed to its publication in an open access journal, and also to require the patient to sign our consent form, which should then be filed in case notes, but not sent to the journal. The authors did so and the manuscript went out to peer review. It was later rejected on scientific grounds. The editors discussed changing journal policy in light of this case to require that authors document, for such papers, that patients have seen the submitted paper and agreed that it could be published, and to cease requiring that authors send copies of consent forms which had been signed by patients. It was agreed to change policy along these lines and the changes are currently being made to our instructions for authors.
A manuscript was submitted about prenatal diagnosis of a specific cardiac disease. Short case reports of four babies who died were included. Although there are no names, we believe that there are sufficient details about the particular baby being described to be identified by the parents and by those involved in their care. The authors argue that:
“As these deaths occurred over 13 years in a large region and the reports only include those with a very specific cardiac diagnosis and do not include any identifying details, we felt it was fair to say that the case reports do not identify the individual. It did not seem appropriate to contact bereaved parents many years later to seek consent for this and we agreed that this was not necessary. The congenital anomalies database has MREC approval through the British Isles Network of Congenital Anomaly Registers. It has also been granted exemption from Section 60 of the Health and Social Care Act 2001 by the Patient Information Advisory Group (PIAG 2-08(e)/2002). This allows us to collect and use the information without specific consent and we believe this covers the use of these case histories.”
Clearly the authors are pleading that they should be allowed to use these case histories on the basis of the best interests of the community.
Is the blanket ethical approval sufficient for us to publish these short case histories, or do the authors need permission from the parents of the children who died?
The Forum’s view was that it is for the editor to decide whether or not he considers that the ethics approval, as obtained in this case, was sufficient. The editor is not bound by the decision of the ethics committee. Many journals take the view that if there is any possibility that the case can be identified, then consent is necessary, and will not publish case reports if consent from the patients or parents is not obtained. If it is impossible to obtain consent, the editor may decide that it is in the public interest to publish the case, but he or she would have to be prepared to justifiy their decision. The Forum questioned the relevance of the case reports and whether the cases could be summarised, omitting all case details. The general consensus was the it was up to the editor to make the final decision and that he should consider the relevance of publishing all of the details of the cases rather than just a summary.
The editors of a scientific journal were sent a letter of complaint from Drs A and B who noticed that a paper had been published online ahead of the print edition authored by Dr C.
Their primary complaint was that they were not included in the authorship and should have been. Other points made in their (rather confusing letters) were that: they had contributed to the paper in the sense that Dr C had used a clinical database which Drs A and B had created and which was at the heart of the research; they had previously asked Dr C to wait before publishing the paper until they had published another paper on a related issue (the “mother paper” on methodology, but this had been delayed by a number of years due to sickness); and Dr A had been the line manager of Dr C (although Dr A has now left the institution). Dr A held the IRB. Dr A had instructed Dr C not to publish until the mother paper was completed.
Dr C was asked by the editors to comment on these points, and he responded by saying that: the intellectual contribution from Drs A and B was minimal; he (Dr C) had added the relevant addendum to the IRB myself; Drs A and B had threatened to ruin his career if he did publish; Drs A and B were preventing publication of the paper by insisting that the mother paper be published first, and yet showed no signs of completing this mother paper; and Drs A and B had now left the institute and the database belongs to the institute. The institute research director (Dr D) knew about the issue and sympathised with Dr C. Dr C was still working at the institute.
The editors felt that it was clear that relationships between Drs A and B and Dr C were poor, and a number of subsidiary points had been made in the letters. However, the journal considered that the issue revolved essentially around whether or not Drs A and B met the criteria for authorship.
The journal website clearly states that the journal follows the guidelines of the International Committee of Medical Journal Editors regarding criteria for authorship.
The journal took the following course of action
(a) The editors wrote to Drs A, B and C and asked first that all three liaise with each other to see if they could come to a consensual agreement about authorship. No agreement was reached.
(b) The editors then wrote to Dr D, the new research director of the institute, and ascertained that the database did indeed belong to the institute. Dr D was asked, as research director, whether he would be prepared to adjudicate on the issue if necessary and he agreed in principle to do so.
(c) The editors then wrote to Drs A, B and C explaining the dispute as they saw it. They stated that they understood that Drs A, B and C believe that they should have been included as co-authors on this paper. That is, they believe that they made substantive intellectual contributions to Dr C’s research study. As authors, they would need to approve the submission of the manuscript to the journal. The editors emphasised their journal’s adherence to the guidelines of the International Committee of Medical Journal Editors regarding criteria for authorship, which are contained in the Instructions for Authors. The guidelines state that the author list should include those individuals who have made substantial intellectual/conceptual contributions to the work. Such contributions involve participation in: (a) experimental design, data acquisition, and analysis and interpretation of data; (b) drafting and/or critically revising the article with respect to intellectual content; and (c) final approval of the manuscript version to be published.
The editors of the journal told the authors that they would adopt the following procedures in resolving this issue:
(1) Given the guidelines above, the editors hoped that the authors might reach an agreement among themselves as to the appropriateness of co-authorship for Drs A and B. Such an agreement would be the best option for resolution. The authors were given a deadline of two weeks to resolve their disagreement on their own, and to inform the journal of the agreement reached.
(2) The editors informed that authors that if an agreement could not be reached on their own, the journal would like to seek mediation from a third party. The editors proposed asking Dr D to make a decision about authorship on the paper in question but noted that all three authors would need to agree to this process, and to abide by Dr D’s determination. Again, the authors were asked to inform the editors of their decision within two weeks (assuming option 1 was not possible).
(3) The authors were told that if they could not reach agreement among themselves, and did not accept the alternative proposal, the journal reserves the right to make the determination itself, from the input provided in the authors’ letters and after discussion with Dr D. The authors were told that the journal would proceed with this third option if any one of the authors informs the journal that neither option 1 nor option 2 is acceptable, or if a response was not received within two weeks.
The issue is ongoing. The editors would be interested in COPE’s view on this dispute and the suggested remedial actions. The editors would be interested to know COPE’s advice in relation to this dispute and how COPE would have attempted to resolve the issue.
Members of the Forum congratulated the editors on their excellent handling of this case. The steps taken by the editors were applauded by the Forum, although the deadline of 2 weeks was thought to be a little strict and perhaps 4 weeks might have been more reasonable. Also, it was felt that the editors should not make the final decision themselves as it is not the job of editors to adjudicate in such matters. In the event that the case cannot be resolved, the advice was to publish the letters from both parties with a statement from the editors.
At the meeting, the following update was provided by the editors. The case has now been resolved. Dr C has agreed to add Drs A and B to the paper. The editors removed the original paper (with 1 author) from the website. An abstract had been published previously on the website with the names of the 3 authors. The editors are planning on publishing the new paper which will have the names of all 3 authors.
Members of the Forum were a little dismayed that the original paper had been removed from the website, especially as the abstract had already been published. When the new paper is published, the advice was to add a note from the editors, as the paper will have two DOI numbers. The editors were also advised to contact PubMed in relation to the fact that there are two DOI numbers for the same paper. The Forum also cautioned that the editors should ensure that Drs A and B sign the author forms before the new paper is published.
A letter was sent to the editor indicating that three articles (one of them in the editor’s journal) on identical subjects had been published in the same year (2006) by the same authors, accusing the first author of all three articles of stealing data from and plagiarising a previously published article from the academic institution where the first author previously worked. The letter, sent by a senior academic and former supervisor of the first author, said that the data had been published without his permission or acknowledgement and he requested that the author be contacted and he and his colleagues punished for their unethical behaviour.
Soon after, the same allegedly victimised head of department sent a second letter to the same editor saying that he was planning on contacting the author’s university and Ministry of Health, informing them of this matter, and also requesting that these papers be immediately retracted from the journal, with an explanation published. Moreover, he stated that he would be submitting a letter to the editor which he hoped would be published with an editorial comment in the next issue of the editor’s journal.
On close inspection of the initial paper published in 2005, referred to by the two letters (the plaintiff was the first author and the alleged culprit the second author), it was clear that the data constituting the only original part of the three 2006 articles were published previously and had been reproduced in identical format in two and differently in the third article. The rest of the data were comparisons with previously published data sets from other countries. The “original” data set had indeed been plagiarised. In addition, the description of the methods was verbatim from the 2005 article, except for the instrument used in all three articles. As a result, there appeared to be clear plagiarism, illegitimate use of data and duplication of data. None of the 2006 articles cited each other and the initial 2005 article was cited in only two of the three articles.
The announced letter to the editor was indeed submitted but rejected on the grounds that informing the readership and issues of sanctions are the prerogative of the editor, not of scientists external to the journal.
The following actions were taken:
The editors of the two other journals involved were contacted suggesting coordination of effort and, if possible, reaching a consensus as to the sanctions. Only one editor replied suggesting: (i) retraction of two of the three articles without indicating which; (ii) contacting the accused scientist and requesting a response to this apparent violation of ethical standards; and (iii) the senior and corresponding authors be restricted from publishing in these journals for a specified period of time.
This editor replied to his colleague suggesting that all three articles be retracted on the basis that none of them provided original data; to simultaneously publish a common letter in all three journals; and lastly to ban the main author for five years and the co-authors for three years.
Questions to COPE: (i) should the current academic institution of the main author be contacted for information? (ii) should there be any dissemination of this case beyond the three journals directly involved? (iii) is attempts to coordinate between journals a good idea or should each journal go on its own and make its own, probably different, decisions?
The editor updated the COPE Forum with the following information. The editor had written to the first author requesting that he retract the paper. The author agreed and his letter of retraction was published together with an editorial from the editor outlining the issues. The paper was retracted.
Members of the Forum questioned whether this was in fact a case of plagiarism, and argued that duplicate publication might be a more accurate description. However, the Forum were unanimous in recommending contacting the academic institution of the main author. The Forum emphasised that the editor should not make any allegations and should not get involved in an investigation except to provide factual information. The institution should be presented with the facts and the details of the 2 other titles, and then asked to investigate the matter themselves. The Forum found the attitudes of the editors of the other journals very worrying. The editor was right in contacting them, but their responses, or lack of response, were disheartening. The advice was to consider contacting the editorial boards of these journals.
The Forum also noted COPE’s usual recommendation of caution in applying sanctions against authors where there has been no due process of investigation, such as an institutional finding of misconduct.
No further action was taken. The editor considers the case now closed.
A group in a developed country performed an audit of pregnancy and its outcome in a group of 250 women with congenital heart disease. There were four maternal deaths of women in the series. It was decided to write up the audit. The paper reports aggregated statistics classified by diagnosis, but also contains a brief account of the factors associated with the four deaths (an account of one of these deaths has already been published in considerably more detail in the confidential enquiry into maternal deaths). Because of the unusual and complex nature of the lesions, these cases will probably be identifiable to medical staff involved in their care, and to close family members. Permission for publication cannot be obtained from the patients, for obvious reasons.
Do the committee consider that the local ethics committee should review the paper before it is submitted for publication? The policy of the local committee specifically excludes audits from its remit.
This case provoked a lively discussion on the legal and moral rights of the dead. Although legally the right to privacy dies with the patient, the committee felt that morally, consent from the next of kin should be sought. In this case it would seem that there is a strong public interest in publishing the cases in an attempt to prevent future avoidable deaths. Balancing the ethical considerations against the public interest, the majority of the committee thought that the audit should be published if it were impractical to obtain consent from the next of kin.
As recommended by the COPE committee, the paper has been referred to the local ethics committee for their comments. The journal is awaiting their comments before proceeding.
The journal was unable to follow-up the case and it is now considered closed.
The journal received a case report for a patient presenting with a particular syndrome in which patients give approximate answers to simple questions. This syndrome has been considered as a dissociative condition but others have argued that it reflects simulation of psychiatric symptoms. The case report was an individual who had crashed his car and, following that, developed complaints of memory impairment and at psychiatric review this syndrome was diagnosed. The patient was reviewed by the authors three years later and showed a number of inconsistencies on examination. Furthermore, it was discovered that, despite the fact that he was unable to recall the day of the week or his own birthday date, he had successfully trained as an official sports umpire. The case thus provided good evidence that this syndrome may be associated with malingering.
The report was reviewed by the Editorial Committee and publication offered as a letter. It was noted, however, that consent had not been provided and this was sought from the authors. The authors explained that this was not available and the Editor explored the possibilities of publishing without patient’s consent, applying the
whether it was in the public interest to publish
whether all attempts had been made to anonymise
that attempts had been made to contact the patient
one could assume that the patient would provide consent if it had been possible to contact him.
It was certainly felt that the paper was sufficiently novel to be published but, despite attempts to anonymise, the Editor had concerns that the individual could be identified. Moreover, the authors indicated that they had not approached the patient as they felt that he would almost certainly refuse consent to publication as the authors had established that he was malingering.
The Editor felt that since it could be assumed that the patient would not consent to publication, then the paper would need to be withrawn. The authors pointed out that this would inevitably produce a publication bias as patients who simulated disease would rarely ever give consent; a view that the Editor certainly shared.
Was this the correct decision not to publish?
The authors have withdrawn their paper, pending discussion at COPE.
While it was recognised that a case such as this, reporting for the first time an important finding, should be published for the greater good, any decision needs to be balanced against the possible harm to the subject of the case report if he or she is identified. It was suggested that one way around the problem would be to remove all forms of identification, including the names of the authors and their institution, but this would present problems in terms of ensuring an available audit trail. An editorial on the subject might be an option, but without using any of the data this would be difficult to produce. A more general editorial on the problems of lack of patient consent in certain circumstances such as in this case where individuals feigned disease, and how the failure to obtain consent could result in publication bias, would certainly be worthwhile.
It was generally agreed that the editor made a reasonable decision not to publish without the patient’s consent but it was a shame that he had to make that decision.
The editor fed back the views of the committee and the paper was withdrawn.
The case of a patient with unresolved upper abdominal pain and weight loss was written up and submitted by her family doctor to a journal that publishes interactive case reports. The intention was to present it as an unfolding story in three parts over five weeks. Responses would be invited on the journal’s website from readers to questions about diagnosis and management, and about what to say to the patient. Several expert commentaries would be commissioned to accompany the third part of the case history. Patients are always asked to give written consent to publication. And the authors are asked to explain that the case will be discussed “live” on the journal’s website and to invite the patient to read the rapid responses and write a commentary about their health and experience of having their story discussed in this way. To date, the patient’s real first and family names have been used in these case reports, with their consent, and they have described patients whose problems have been resolved. Would it therefore be ethical to publish this unresolved case, given that the patient may be exposed to all sorts of opinions on her health, some of which may be very frightening? Can consent in this situation be truly informed?
- There is a duty of care to safeguard the patient’s psychological health. - The potential harm is completely unpredictable. - Why is the patient named? This will only increase the possibility of harm. - This is tantamount to “reality medicine,” akin to a Victorian freak show. - The patient should not take part as it is not in their best interests. - It would be difficult to obtain consent in this case, as it is a work in progress with huge risks.
Some authors tested the effect of a food on the menstrual cycle. The manuscript included patient identifiable information, but the authors did not provide formal confirmation that the patients consented to publication of the study. Information was sent to the corresponding author, outlining legal obligations in respect of patients' consent to publication. But the authors stated that they considered themselves exempt from these regulations because they are research scientists and not healthcare providers. Furthermore, under the guidelines of their university's privacy office, all personal information relating to study subjects must remain confidential. To provide the journal with the appropriate authorisation from the patients would therefore contravene these guidelines. Under certain circumstances, the privacy review board may waive authorisation and allow publication without explicit consent to publish from patients. - Should the regulations apply equally to healthcare providers and research scientists? - In what circumstances would the privacy review board's waiver of authorisation be acceptable?
- The journal should set guidelines as to the standard of consent to publication that it required. To publish, authors would have to meet this standard. - These regulations do not allow for named consent, and they work prospectively in that patients give their consent to any future publication. The publisher must then keep that information confidential. - Were the subjects of the study patients or healthy volunteers as there is a different framework if the research subjects are not patients? - Ultimately, it is not possible to provide an answer to the editor’s questions. - The editor should contact the agency responsible for administering the regulations.
A paper described a problem of two women who wanted their fetuses to be tested for a genetic condition, but where in both cases their partners had refused to give consent. Should the journal publish such a paper without obtaining consent from the partners? The editors think not, but the authors are unconvinced.
_ It would be impossible to completely anonymise the case even if mention was not made of the genetic condition. _ One solution might be for the case to be published in the form of a discussion and alluded to in an abstract sense, although the authors had indicated that they would prefer to leave the case reports in. _ Public interest can override confidentiality.
The journal published the article on the general subject, but with no information on the particular cases. The authors eventually agreed that it would be wrong to publish the case reports without consent from the partners.