Open-access electronic case report journals: the rationale for case report guidelines.

Case reports serve three primary functions: generating hypotheses for verification in subsequent longitudinal observational studies and clinical trials, documenting rare or unusual phenomena where obtaining further evidence is not feasible, and providing clinical stories that help health care providers remember important lessons. Vandenbroucke [1] previously discussed these roles in depth, describing how the use of deduction, induction, and serendipity could produce case reports that lead to more formal investigations, case report strength of evidence, and the aggregation of multiple case series into meta-analyses to generate meaningful conclusions. The latter two observations increasingly have been viewed with skepticism, partly because of the rise of evidence-based medicine (EBM) in the early 1990s. EBM strives to integrate the best available published research with both clinical expertise and patient values and circumstances [2,3]. Case report findings are not generalizable, do not address causal inference or explanatory mechanisms, and emphasize low-probability events [4,5]. Systematic reviews or meta-analyses of multiple case reports or case series add numerical weight, which can improve the chances of detecting unusual associations. However, pooling data does not in itself allow a rigorous assessment of causation when the source material is composed solely of multiple case reports lacking longitudinal data. In addition, sufficiently complex diseases may have heterogeneous clinical presentations. Simply pooling cases that ostensibly represent the “same” disease can sacrifice sensitivity to increase specificity, while in the process deemphasizing salient details of each case [6]. The EBM movement consequently tends to categorize case reports as weak evidence, and we agree to an extent. However, that does not mean that they are of little importance. Their role in uncovering important phenomena originates in the ancient past: Hippocrates’ Aphorisms, the Edwin Smith Papyrus, and the Yellow Emperor’s Classic of Medicine are all compilations of case reports and anecdotes that are among the first written documentation of medical concerns [7]. In contemporary times, the first article of the inaugural issue of the New England Journal of Medicine 200 years ago was a descriptive treatise on a series of patients with angina pectoris [8], and five of the 51 articles in JAMA’s 1985 centennial compendium Landmark Articles in Medicine were case reports [9]. The educational value of well-written case reports is also underappreciated. The human brain is particularly good at learning and remembering through examples and anecdotes, and case reports can offer valuable and memorable examples of how a disease might present or how to avoid certain complications when doing new procedures [1,7,10,11]. Health care providers can benefit greatly from discussing patient stories, which can be more effective and inspiring than impersonal statistical abstractions. Recognizing that medicine is as much a story-based narrative process as a scientific one can enhance empathy with patients and provide perspective [12]. For addressing clinical conundrums, case reports can help generate hypotheses, highlight ethical pitfalls, and bring to mind diagnoses that health care providers might have otherwise overlooked. Herein, we will discuss two additional phenomena that have further obscured the already debated role of case reporting in modern medical practice. First, the recent rapid growth of open-access online-only journals focused on case reports has lowered authorship and editorial barriers to publishing. Second, the continued lack of publishing guidelines for case reports has led to variation in quality and thematic presentation across journals and clinical disciplines. Consequently, clinicians intending to incorporate useful findings from case reports into their practices face a daunting challenge. We outline current case report publication trends in the later section and provide suggestions for quality improvement in case report writing and editing that may help strengthen their clinical, scientific, and educational value for health care providers.