We received a paper which describes genotyping results from a large number of individuals (>50) from five unrelated families, in which family members had various blood and liver conditions. On submission we noted that the paper included specific details regarding the clinical histories of individuals in each family. Some individuals were described in substantial detail, others only briefly. For example, information about probands included age at presentation, sex, ethnicity, clinical history, occupation, clinical complications (some quite specific), clinic attended (for some individuals), history of alcohol consumption, ages of relatives, clinical details for relatives, age at death etc.
The authors responded that when initially obtaining consent to the research project, patients consented that “[t]he results from studies on the research samples may be published, but individual patients will not be identified in the publications”. They claim that details included are not identifying, and that it would be impractical now to trace all living relatives. They asked whether our consent form has been approved by an IRB, and say that before using it they will need to have it reviewed by their IRB.
We are unsure how to proceed but feel that when consenting to the research project the individuals may not have realised they would have been described to this level of detail, and that we should respect their privacy. One option might be to ensure the authors seek consent to publication from all probands, but then remove extraneous detail regarding the relatives. However, it is possible that the IRB may have useful input regarding the publication of individuals’ details from this study.
The editor was asked if the personal information was essential to the study. If it were deleted, would the paper still have value? The editor confirmed that the information was crucial to the study and could not be deleted. All agreed that the editor needs to weigh up the need to publish against any harm that might be caused by publication. Most believed that the harm involved was probably quite severe and so outweighs the need to publish. One test that can be applied is to consider whetheran investigative journalist would be able to identify the patients from the study (in this case, most Forum members felt that they could, therefore the risk of identification is high). The Forum did not believe it was that impractical for the authors to obtain consent and so they thought it was reasonable to ask the authors again to obtain consent. Did the authors make it clear to the patients how much detail and history would be published? Also, some noted that with pedigree studies, it can be very easy to identify patients. As with all such cases, the editor needs to make a judgement concerning the harm that might be caused to the patients in publishing and the benefits of publishing. Most believed that the risk of identification was high and that the editor should not publish without consent. The advice was to go back to the authors and ask them to obtain consent. COPE was asked about whether it could produce a generic consent form for all journals (to avoid problems of having to use multiple forms if a manuscript is reject by one journal and submitted to another). COPE Council agreed to consider this suggestion.
The editor informed the authors that the case had been taken to COPE, and the agreement of those who attended was that patients and family members would need to give consent to publication of the case(s) having read the paper. The authors responded after several weeks to say they had traced the patients and obtained consent to publication. They updated their paper to state this in the revised text, and also updated the text to minimise some details of family members, and to state that details were minimised to maintain anonymity but that further clinical details could be made available to other researchers on request.
We recently published article A by author group X on our website ahead of print publication and subsequently received a formal complaint from author group Y alleging that the paper constitutes a breach of their intellectual property rights.
Group Y state that the described work is based on a jointly developed concept, initially resulting in a joint report (published 2004). In their view, article A therefore contains their intellectual property, which is documented by frequent email exchanges, delivery of all relevant reagents and two visits by the senior author in group X to group Y's laboratory.
Group Y further state that there has been a previous unauthorised publication of a report that also contained their unacknowledged input (intellectual input and reagents), resulting in group Y prohibiting any further publication on the same topic by group X without their authorisation (!?).
In article A, the authors state that they used commercial reagents, although group Y allege that their reagents were available to group X at this time. Group Y are seeking what they perceive to be correct acknowledgement for their contribution, and there is an ongoing legal dispute, which they are threatening to expand to include article A unless a suitable solution is found.
We approached group Y to clarify if they thought they should be acknowledged on the paper, or even included as authors, but they did not answer, as they believe that group X should be asked to clarify their position.
We approached group X for their response to these allegations. Group X maintain that their collaboration with group Y was their idea, as an obvious extension to their previous clinical publications. Group Y are chemists and had not published on any clinical topic prior to the collaboration with group X. Group X state that the collaboration came to an end in 2006 when the senior collaborator in group Y demanded that either s/he should be named as the last author or one of the junior members of group Y named as first author on any manuscript resulting from the collaboration. Group X considered this to be unreasonable and continued their studies using commercial reagents.
The editors of the journal in question believe group X's response to be appropriate and complete, and find no grounds for taking group Y's complaint any further. The editors would like to proceed with print publication of article A.
Is proceeding with publication at this stage appropriate? The editors are concerned about the threat of legal proceedings, so should they therefore be more cautious in accepting one version of events above another—do they need to request “proof” and, if so, what would this proof consist of? The Forum’s advice on the next steps that they should take would be appreciated.
The Forum agreed that the paper should be considered published. It is irrelevant whether this is online or in print. Although one suggestion was to let group Y have their say, perhaps in a letter to the editor, others cautioned against this approach in the light of unresolved legal issues. If legal proceedings are threatened, the editor should withdraw from the dispute and not investigate the matter further. The response from group X could be shared with group Y only after explicit consent from group X. The advice of the Forum was to go ahead with the print publication of the paper but the editor should not get involved in correspondence between the two groups and should seek legal advice.
The Editor took COPE’s advice and no further action resulted. The editor considers the case now closed.
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.
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.
A journal provisionally accepted a case report. When requested, the authors could not identify patients to obtain their signed consent. The authors offered to anonymise the data, but the journal was inclined to decline. - What should the editor do?
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.