Guidance for Writing Case Reports in Addiction Medicine

Methodologically sound interventional and observational studies contribute to knowledge of prognosis, efficacy, effectiveness, and risk. Evidence pyramids place the highest value on systematic reviews and meta-analyses of such studies.1 These studies do provide generalizable results in populations that can be applied to individuals. But they do not reflect the richness and complexity of individual patient experiences or cases in their contexts. Case reports, although they cannot provide conclusive evidence for efficacy or risk, can provide valuable information of a different type that can impact medicine across several domains. Case reports: (1) can foster clinical reasoning, (2) are feasible scholarship for many, (3) can serve as an alert to hazards, (4) can signal new clinical and service delivery practices, and (5) inform health policy. In medical education, grappling with real cases is a long-lasting way to foster clinical reasoning among trainees in accordance with adult learning theory,2 so cases often form the bases of grand rounds presentations and standardized test questions. Case reports also are considered a form of practical scholarship for those in training and clinical practice who have frequent occasions to make direct clinical observations, and opportunities for their publication have grown exponentially in recent years.3 These scholarly reports can alert clinicians and public health agencies to health hazards beyond the reach of clinical trials—eg, vaping and lung injury4 or the harms of synthetic cannabinoids5—and to signals of possibly efficacious new practices that should be tested in more rigorously designed studies.6,7 They can serve as stimuli for clinical and systems improvement by highlighting challenges and pitfalls in current practice.8 In health policy, cases capture the minds of voters and lawmakers, informing the allocation of resources based on a combination of facts and the emotional tug of storytelling. Consider Matthew Casey Wethington, who died in 2002 from a heroin overdose at the age of 23, whose story—and the advocacy of his parents—led to Casey's Law in Kentucky that supports involuntary treatment of people with substance use disorders.9 It is no surprise, then, that cases can play a particularly important role in teaching, studying, generating new knowledge, and advocating for change in addiction medicine, a field whose context is rapidly evolving. Our experiences editing case report submissions to the Journal of Addiction Medicine have reaffirmed to us the important contributions of case reports to the field of addiction medicine and have also alerted us to their challenges and hazards. Our purpose in this editorial is to identify 4 potential pitfalls of case reports in addiction medicine, and to offer guidance for authors considering writing and publishing case reports in addiction medicine and related fields. The first potential pitfall emerges from a relative lack of expertise and established standards in case report writing compared to the writing of other scientific articles, for which norms of accuracy, completion, and transparency of reporting have been compiled.10 The available reviewers for a journal, as well as its editors, may have less experience with case reports than other types of reports. Instruments to assess rigor in case reports are generally lacking, whereas other research approaches such as randomized trials or cohort studies can be systematically assessed for bias and quality via tools like the Cochrane Risk of Bias Tool11 and the Newcastle-Ottawa Scale,12 respectively. Such reporting standards ensure completeness and transparency of published reports and facilitate their systematic aggregation. The second potential pitfall emerges from the sometimes murky distinction between case reports and human subjects research, particularly in scenarios where a new treatment is tried by one or a few patients. How ought consent be obtained in case reports, and when is ethical review (eg, by an institutional review board [IRB] or ethics committee) required? Consent to be the subject of a published case report and consent to participate in a research study are different processes with different guidelines, and authors of case reports may not clearly identify to exactly what the subject(s) consented. A third pitfall involves a lack of enough information in the report to reliably make substance use disorder diagnoses, assess change over time, or claim effect of an intervention. Internal validity in case reports depends on the triangulation of multiple forms of data, such as clinical history, physical exam findings, and biochemical testing.13 Given the care fragmentation experienced by many people with addiction and other comorbid conditions,14,15 whereby primary care services, addiction services, and the management of other conditions may occur in different institutions, data triangulation can be logistically difficult for authors of case reports just as it is for clinicians. Data triangulation can be further complicated by federal regulations specific to addiction medicine, such as Title 42 of the Code of Federal Regulations Part 2, which controls the release of patient information about treatment for substance use disorders.16 Furthermore, patients with substance use disorders are subject to overlapping forms of stigma, discrimination, and power relations with health care providers17, which may impact their reachability by clinicians seeking to verify or collect additional information for the construction of a case report. A final pitfall is a structural and stylistic one. Unlike other types of scientific articles, case reports do not have specific aims or hypotheses so readers searching for a “take-home point” may not find one as easily as they might while reading other articles. Therefore, without clear statements of the reason for reporting the case, and efforts towards contextualizing the report in the medical literature, it can be unclear to editors and to readers what is novel about the report and how it should inform readers’ thinking about a clinical or systems problem. Bearing in mind the unique potential pitfalls and strengths for case reports in addiction medicine, we propose the following ethical, reporting, and writing guidance for those writing and publishing them (Table 1).TABLE 1: Specific Guidance for Writing Case Reports in Addiction Medicine1. Obtain consent and state clearly in the report what was obtained (eg, written consent was obtained from the patient to report their case in the medical literature). Accepted standards of consent, such as those supported by the International Committee of Medical Journal Editors (ICMJE), should be adhered to.18 Consent guidelines specific to case reports, such as those presented by the Committee on Publication Ethics (COPE), can guide writers.19 While individual journals may vary in their guidance to authors regarding the format in which consent should be obtained, the ICMJE and COPE generally recommend written consent to report the case and that the subjects of the report approve the final manuscript if any potentially identifying information is presented. Authors of case reports need not consider consent to be a one-off, but an ongoing process that may include multiple points of contact between the writers of the case report and its subject(s).20 At the least, agreement between subject(s) and author(s) as to the decision to write the report as well as approval of its submission should be documented in the report. More than consent, seeking feedback from patients during manuscript preparation can raise the voices of marginalized persons and help disseminate their perspectives.21,22 In cases where consent cannot be obtained, reasonable efforts in multiple modalities (such as in-person outreach, phone, internet search, or mail) to obtain consent should be documented in the report. In these situations, the authors should explain their reasoning for not obtaining consent and their efforts to do so, they should minimize identifiable features in the report, and the authors (and editors) should attempt to balance the risk of deductive disclosure with the benefit of the case report to public health and science. For example, an identifiable case report (with associated disclosure risks) with little impactful content should not be published (without consent). An anonymous case that has great value could be published without consent after extraordinary efforts to obtain consent failed. 2. Obtain IRB/ethics committee review if appropriate. Authors should reflect on whether their project constitutes human subjects research, and if it may, ethics review by an IRB should be performed and documented in the report. While consent to report a case and consent to participate in research are not the same, the former is always needed for case reports. The latter is needed when there has been human subjects research that requires consent. IRB review is required when the activity is human subjects research, whose definition is widely accepted to include any research involving a living individual about whom data (including private information) or biospecimens are obtained systematically for research purposes to generate new, generalizable knowledge.23 Therefore, a trial of a new treatment in a single patient with such intent would require review by an IRB. However, a report of a clinical case, in which no information was collected solely for research purposes but was instead done so for clinical care, and no new therapy was trialed, would not. When more than one patient is the focus of the report (which thus constitutes a case series), IRB review is more strongly encouraged. Several major research institutions have concluded that a case series involving greater than three patients ought to undergo IRB review because they are more likely to meet the definition of research as a systematic investigation.23 For the sole benefit of a patient, clinicians sometimes try new treatments. Such cases might be reported retrospectively as case reports if the initial intent did not include systematic evaluation to generate generalizable knowledge. However, to avoid any confusion regarding whether a new treatment is research or not, we strongly encourage IRB review and approval of any new treatments delivered whose results might be reported with the intent to generalize the findings in a case report. Such new treatments for clinical purposes may also come under Food and Drug Administration (FDA) “single patient expanded access” or “emergency use” regulations for which there are specific IRB and FDA procedures.24 When drugs are used off-label or with novel dosing strategies in a patient for whom clinicians prospectively decide to collect and report outcomes, these clinicians should consult the FDA's guidelines regarding whether an investigational new drug application (IND) is warranted.25 3. Triangulate data from multiple sources to minimize threats to internal validity.26 For example, substance use can be documented by history as well as by body fluid testing, and substance use disorders and their severity can be diagnosed based on criteria established by the Diagnostic and Statistical Manual, 5th revision.27 Authors should also state how they assessed for diagnostic criteria, for example, via checklists or other instruments, structured interview, and/or a clinical examination. Case reports in addiction medicine that do not include laboratory testing of substances often have limited value. 4. Characterize the relationship between author(s) and subject(s) to clarify potential bias and allow readers to interpret power relationships between writers and subjects.26 Who solicited historical information, and who obtained informed consent from the patient and at which time periods during the unfolding of the case should be clear to readers so they may determine if internal validity is threatened by bias related to social desirability or coercion. 5. Use reporting standards and structure the report. Cite the standard(s) to which the report adheres. We suggest using internationally accepted reporting standards for case reports.28 Corresponding checklists ought to be made available to editors on request. Case reports should have an introduction, the report itself, and a discussion of the case's implications. Introductions to case reports can offer up-to-date, concise reviews of the literature that educates readers about the topic and justifies the need for sharing the case. The justification may address the novelty or uniqueness of the case (eg, a rare disease or rare presentation) or its relationship to a contemporary phenomenon (eg, a success or failure of a feature of a care system, or demonstration of how to apply evidence from a clinical trial). After the case is presented, a discussion should follow that integrates the case in the larger context of what is known on the topic. If hypotheses are generated by the case, they may represent opportunities for future research and these should be stated. In stating a “take-home point,” authors should avoid drawing firm conclusions regarding efficacy or harm given the methodological limitations of the case report. Limitations in drawing conclusions and threats to internal validity should be stated clearly for readers. Strengths should also be noted (eg, the use of biological fluid testing). The discussion section may also include a perspective from the patient(s). 6. Use non-stigmatizing, person-centered language so that the case report may reduce, and not perpetuate, the stigma embodied in certain outdated terms (eg, addict, abuse) that can influence providers’ attitudes and behaviors.29 Authors are encouraged to reference the statement made by the International Society of Addiction Journal Editors that refers to the destigmatization of language surrounding addiction.30 William Osler, a foundational figure in clinical practice and medical education, is credited with many famous pearls, including “the good physician treats the disease; the great physician treats the patient who has the disease.” Case reports shift our gaze from the disease to the patient, and in so doing, can help identify novel disease presentations, successes and failures of health systems, and share the experiences of vulnerable patients. As the field of addiction medicine—as well as its legal, cultural, and policy milieu—evolve, case reports represent an opportunity to inform clinical practice, research, and policy. We hope that those who study and treat patients with substance use disorders will take advantage of this form of inquiry and dissemination of knowledge and, in so doing, will encourage readers to take more thoughtful and person-centered approaches to addiction medicine and related fields.

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