Patient inclusion in case reports: a call to action

A case report presents an account of an individual patient's clinical course. Information on diagnosis, treatment and follow-up is usually presented alongside learning points and links to relevant literature. A hierarchy of evidence-based medicine exists, with case reports forming the lowest level of clinical evidence [1]. Nevertheless, good quality case reports remain a useful source of information, can capture and describe important scientific and clinical observations concisely [2], and can provide important patient-centred insights. An intrinsic part of the definition of a case report is the focus on the individual and the learning points that can be taken from it, for example the diagnosis or management aspects. A case report should ideally be a holistic account, which might be one-sided if the patient's voice were absent. Rather than this being merely an aspiration, our opinion is that every effort should be made to include the patient perspective to give weight to the patient experience. To accomplish this, there must be a willingness amongst clinicians to give the patient a voice. If authors do not actively seek patients' accounts and are not prepared to share this experience, a crucial part of the evidence is lost. The World Health Organization (WHO) recognises that patients, families and other informal caregivers bring insights from their experiences of care that cannot be substituted or replicated by clinicians, managers or researchers [3]. Patient-reported outcome measures (PROMS) have been collected since 2009 for NHS patients undergoing certain procedures such as hip replacement and surgery for the repair of groin hernias and varicose veins. However, until the Patient Library was set up in 2015, there was no national evidence base for patient experience [4, 5]. In the Inadmissible Evidence report of the Patient Library [6], an NHS-wide failure to take evidence of patient experience seriously enough is described, with greater weighting typically attributed to the reports of medical professionals when compared to those of patients. A contributing factor is the language used by medical professionals to describe evidence of patient experience. When a health professional flags something that has gone wrong, it is called an incident report. When a patient does the same, it is called a complaint, a word which invokes ‘objection’ or ‘grievance’, and contrasts with the apparent objectivity of an incident report. Aside from adverse incidents, a similar language problem affects more general patient feedback, for example that gathered via the Friends and Family Test [7], patient surveys, and focus groups which - all frequently described as ‘anecdotal evidence’. The term indicates a tendency to see patient feedback as subjective, irrational, and potentially unreliable [6]. Contrasting ‘incident reports’ to ‘complaints’, ‘hard evidence’ to its ‘soft’ or ‘anecdotal’ counterparts, and professional ‘records’ against patient ‘stories’ suggests a culture in which the weight and importance of some forms evidence are seen as being greater than others. From patient safety groups and reports, similar themes and problems are identified, for example by Baroness Cumberledge in the First Do No Harm report of the Independent Medicines and Medical Devices Safety Review [8]. In recognition of the missed opportunities to learn from patient accounts, the WHO recently stated that, “patient engagement and empowerment is perhaps the most powerful tool to improve patient safety. Patients, families and other informal caregivers bring insights from their experiences of care that cannot be substituted or replicated by clinicians, managers or researchers”. To this end, the WHO advocates “amplifying the patient voice as a force for improvement”, which conveys not only the need to hear, but also to champion and support patient perspectives [3]. Although written from a patient safety standpoint, this move towards normalising the inclusion of the patient perspective is an important positive step, relevant to all aspects of healthcare. Case reports can be an accessible route for a patient and their community to make a contribution to involvement in the published literature. It is recognised that patients who do not understand or speak the language used by researchers, or who have difficulty communicating for any reason are commonly excluded from taking part in clinical studies. These patients often belong to under-served groups [9]. These are individuals currently underrepresented in publications relative to population estimates, for whom the research community needs to provide a better service. Although this is an issue which is becoming more widely recognised and initiatives are being developed to try to broaden access, everyone should be provided with the support required to contribute, to feel that scholarship ‘works’ for them too, and will strive to enable their voices to be heard. The practical barriers to a patient's voice being heard should be considered. Following a significant healthcare-related event (for example, a medical error or major illness), a patient may try to simply to put it behind them and move on. However, they may wish to engage with the healthcare institution or educate the healthcare community, but for members of the public it can be very difficult to do the latter. Many may not have the knowledge, confidence and resources to approach clinicians or those in academic medicine. Whilst there are a few examples of medical literature written by patients (including case reports), these are rare. Anushua Gupta shared her experience of extra-corporeal membrane oxygenation during the COVID-19 pandemic [10], although it should be noted that the author is also a general practitioner. Another example is Susanna Stanford's critical appraisal of a systematic review on inadequate neuraxial anaesthesia during caesarean birth [11]. A more established route for the publication and dissemination of patient experiences is for clinicians to seek, record and report them on the behalf of the patient. It is imperative that the patient gives their informed consent (or assent of a consultee or guardian, as appropriate) prior to the publication of a case report, and the inclusion of the patient's perspective should be recommended in author instructions to emphasise that the patient should be asked if they wish to also have their voice heard [12]. Accurately representing the patient's insights should be a priority for both journals and potential authors and authors may be asked during the submission process to indicate if the patient has actually been consulted and asked to contribute. There are pros and cons including the loss of anonymity which should also be discussed. Unless the patient is a (co-)author of a case report, the patient's name should not be published, and any supporting materials such as photographs or investigations should be de-identified. However, it is not always possible to guarantee complete anonymity. This is especially true now that that publication is often accompanied by dissemination via social media. This enhances the impact of the publication through extending its reach to a wider audience. However, publicity may increase the probability of identification, for example by friends, relatives, or members of their healthcare team. This possibility should also be discussed as part of the consent process, as the possibility that their case may reach the attention of the public rather than just the medical community must similarly be recognised. In all cases, the authors should provide the patient with point of contact in case they wish to withdraw their consent prior to the publication of the report. We suggest that this should be a departmental or institutional contact rather than a specific individual, to ensure that contact can be made as easily and reliably as possible. A well-managed and ethically robust consent process should mitigate the risk of any unexpected breaches of privacy. However, the patient should be informed of where the report is published, so they can contact the journal should this become necessary. They should also be allowed to read the report and see the actual photographs that are being published. Retractions of case reports are rare, but the Committee on Publication Ethics notes that retraction may be appropriate if papers contain “material or data without authorisation for use”, or when there is a “serious legal issue (e.g., libel, privacy)” [13]. Responsibility for retraction of published material lies with the journal rather than the authors, and journal editors should put measures in place to limit negative impacts for patients involved, should a case report breach ethical principles or privacy. Building on the above principles, a robust framework to protect patients who wish to contribute to case reports would be beneficial, and we suggest that this may be best defined through a consensus process involving patients, carers, authors and editors. When seeking to record patient perspectives, open questions should be used to ensure that authors learn from patients rather than directing what they say. Patients should be reassured that their contributions will be valued, to help overcome the patient perception of the hierarchy described above. The authors should be aware that a patient may not be ready for an interview, for example following a significant event and flexible timing is therefore crucial. Whilst not all case reports follow a difficult situation, recent evidence on post-incident support can provide useful guidance for staff on how to support learning and improve their understanding of patients' perspectives [14]. This suggests that access to appropriate senior support is made available after the event, and written communication with the patient's general practitioner may be appropriate [15]. However, a recent review of the evidence on key stakeholder perspectives when patients and families are involved in serious incident investigations indicates significant gaps in the literature [16]. It may be appropriate to talk to the patient soon after the episode of care described, but a time delay before a final version is written may be needed to allow for the patient to reflect. Furthermore, there may be a wider perspective and interactions with other parts of the healthcare system (including non-medical colleagues and those from other departments) may have become relevant, especially as transitions of care can be difficult to navigate. This needs to be acknowledged by the authors who are keen to get published and may overlook this important step in the case report writing process. The value of the patient voice should become more widely accepted and its contribution to knowledge appreciated. Its inclusion in high-quality case reports is an important step towards responding to Cumberledges's assertion that “the patient experience must no longer be considered anecdotal and weighted least in the hierarchy of evidence-based medicine”.