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

https://doi.org/10.1016/j.jclinepi.2013.04.001Get rights and content

Introduction

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.

Section snippets

Case report publication trends

An April 2012, general search for “case report” on PubMed and EMBASE yielded about 1.6 million and 2 million hits, respectively. These represent about 8% of all published records in both databases since their inception [13], [14]. The total number of new articles classified as case reports for each year from 2000 to 2010 is shown in Fig. 1. Annual case report volume increased from 42,439 publications on PubMed and 49,918 on EMBASE with MEDLINE in 2000 to 61,689 and 72,388, respectively, in

The online case report publishing movement: risk vs. reward

BioMed Central and the BMJ Publishing Group (publishers of the influential British Medical Journal) are two institutions that have pioneered the online distribution model for medical publishing, using case reports as the exclusive foundation of new subsidiary journals. The first case report–only journal, Journal of Medical Case Reports, was launched by BioMed Central in January 2007 [16]. In 2008, BioMed Central established a second case report–only periodical, Cases Journal [17]. The same

Suggestions for improving case report standards

We propose that the optimal case report should follow the format of an “old school” Grand Rounds, including putting the case into appropriate perspective from a clinical and/or research audience. If the impetus for the case report is educational, then the evidence supporting it as an illustrative case must be well documented and articulated. We present in the later section a potential checklist for high-quality case reporting, based on suggestions from prior publications [5], [26], [27] and

Further considerations for case report guidelines

If case reports are to be respectable and useful instruments for improving patient care and advancing research, improving their content and presentation quality is important, and we hope that reporting guidelines might be one method of achieving this goal [5], [29]. Although scientific writing per se is not one of the six core competencies of the Accreditation Council for Graduate Medical Education, communication is. As case reports are in many instances simply a written conveyance of a

Acknowledgments

The authors thank Mark P. MacEachern, MLIS, for his assistance obtaining figures for annual case report publication volume in PubMed and EMBASE and Cyrus A. Raji, MD, PhD, for his insights regarding biomedical research trends.

Disclosures: Gordon Sun and Oluseyi Aliu are Robert Wood Johnson Foundation Clinical Scholars supported by the US Department of Veterans Affairs. The Robert Wood Johnson Foundation and the Department of Veterans Affairs were not directly involved in study design, data

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