The Case for the Case Report
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J. Jeffrey Malatack, MD The Case for the Case Report Case reports at one time could be found in many if not most medical journals. I found that 40 years ago, the top 10 pediatric journals all published case reports (many with designated case report sections), and 30% of the articles published in the 10 journals collectively were case reports. Twenty years ago, these same journals had reduced their case report publications, averaging only Pediatrics15% of their published articles, with many having dropped a case report section altogether.Pediatrics In 2016, case reports only accounted for 4% of articles published in this same group of journals, with Pediatrics’ at 11% being the most committed to this format of medical publication. also is the only one of these publications with a designated section for case reports. recent decision to reduce the number of published case reports from a maximum of 8 an issue to 3 an issue is the latest in this long decline. It appears that 2 forces have had a role in the decline of the case report as a genre of medical publications. The 2 forces include the impact factor, created in the 1990s, and the inability to use statistical methodology to assess the data at hand. The impact factor is a measure of the frequency with which the average article in a journal has been cited from a particular year. It is used to measure the importance or rank of a journal by calculating the times its articles are cited. A 2-year period is used and involves dividing the number of times articles that were cited in that 2-year period after publication by the number of articles that are potentially citable. Case reports are handled, for this purpose, as are original articles. Case Reports, for reasons outlined below, are invariably not going to be cited as often as original articles by the 2-year postpublication period that defines the impact factor. “ ’ The other force that has led to the decline of case reports is the inability to apply statistical analysis ” to strengthen the scientific merit of the report. In the words of one of my coeditors, You can t tell ’ anything from a single case. The error in this reasoning is the supposition that a case report and an original article have anything in common. They don t. Pediatrics, Sidney Kimmel Medical School of Thomas Jefferson University and Division of Diagnostic Referral, Department of Pediatrics, Nemours Alfred I. duPont Hospital for Children, Wilmington, Delaware DOI: https:// doi. org/ 10. 1542/ peds. 2018- 0328 Accepted for publication Feb 13, 2018 Address correspondence to J. Jeffrey Malatack, MD, Diagnostic Referral Department, Nemours Alfred I. duPont Hospital for Children, 1600 Rockland Rd, Wilmington, DE 19899. E-mail: [email protected] PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275). Copyright © 2018 by the American Academy of Pediatrics FINANCIAL DISCLOSURE: The author has indicated he has no financial relationships relevant to this article to disclose. FUNDING: No external funding. POTENTIAL CONFLICT OF INTEREST: Dr Malatack is a member of the editorial board of Pediatrics and is the Case Reports section editor. Downloaded from www.aappublications.org/news by guest on September 30, 2021 PEDIATRICS Volume 141, number s5, April 2018:e20180328 SUPPLEMENT ARTICLE WHAT WAS A CASE REPORT DURING ITS Exceptions to a Current Paradigm GOLDEN AGE? yet another study identified the frequency at which a typical kitten Many years ago, we published a Bartonellaharbors the henselae CSD organism that is now Case reports at one time, when many report of cat scratch disease1 (CSD) in called, after multiple7 names changes, more were published, included the absence of adenopathy. At the . The original infrequently seen but well-described time, before the identification of the observation of CSD occurring without diagnoses as reminders of rare putative organism of CSD, the dogma adenopathy remains the discovery conditions one should not miss. was that anyone suffering from CSD published as a case report. It was The authors of other case reports had to have adenopathy. A so-called years after that original observation sine qua non. However, an expert on before the studies mentioned were described unexpected combinations ’ of disease processes as an unlikely CSD, after hearing the details of our published. These studies added to occurrence in a single patient. Still case, including the child s intimate the body of knowledge on CSD and others reported on the diagnosis contact with a kitten and despite the emerged from the paradigm shift of a well-described condition from absence of adenopathy, sent us the after the time it took to develop a a geographical area not having then-only diagnostic test for CSD, a study protocol, collect study data, reported the disease before. skin test (Hanger Rose test) that one2 analyze the data, and then publish These kinds of case reports have applied by an intradermal injection. the findings. Any reference to the 2 all but disappeared from medical Sure enough, the test was positive. original case reports on CSD without To add additional certainty to the lymphadenopathy were never publication. Reminders of rare ’ conditions or unusual presentations diagnosis, we sent the tissue from a credited to an impact factor because ’ of common conditions are no granulomatous lesion in our patient s of the multiyear delay between these ’ longer necessary when the world s liver to Colonel Douglas Wear, MD.3 2 case reports being published and literature is at everyone s fingertips. A few years earlier, Dr Wear et al, Asubsequent Starting Point studies for years Hypothesis later. Coincidence adds nothing to medical while working at the Armed Forces Testing Research knowledge and the world has been Institute of Pathology, had reported so functionally reduced in size that a Warthin-Starry staining organism in the lymph node of a patient isolated areas free of a given medical A case report also can be an with CSD. Dr Wear identified those condition have nearly disappeared. ’ observation that gives life to a novel same silver staining organisms As case report publications have idea (hypothesis). Although ideas in our patient s liver granuloma. dwindled, the definition of what The Lancet derived from a single or even a Coincidentally, this identical finding makes up a publishable case report The few patient interactions might as was published in shortly has evolved into something different. Journal of Pediatrics easily be a misstep down the path before our report appeared in 4 to knowledge, they also can and WHAT IS A CASE REPORT NOW? . The paradigm have been the beginning of novel changed. Since those 2 published understanding. In these instances, reports, no one has performed a case reports must be put to the test double-blind study to confirm the of the scientific method before the A case report at its best is a clever new paradigm. It has never been idea can take hold and be accepted. idea that arises from a physician- necessary. If CSD can occur without Ideas are precious and should patient interaction. The authors may adenopathy, the possible spectrum never be squandered. It is perfectly uncover an exception to the current of CSD became much wider than “ ” acceptable to eliminate ideas that paradigm, raising the proposition previously believed. In the ensuing emerge from case reports that fail of if 1, then some, and as such years, the authors of a number of the test of scientific rigor so that demanding a new paradigm able studies published as original articles those ideas that do emerge from that to incorporate these exceptions. about CSD took advantage of this rigor unscathed can add to medical A case report may be a starting point increasing spectrum of disease. knowledge. Ideas are not science, but for hypothesis-testing research. The The authors of 1 study indicated they are necessary at the beginning authors may suggest a new therapy how fever of unknown origin is 5 of a scientific advance no less than or raise an early alert regarding frequently due to systemic CSD. an acorn is necessary to allow an oak untoward consequences of a new Others identified how frequently a tree with the potential to be a giant, treatment, or they may raise the previously healthy child presenting to burst into existence and begin to practice level of the reader. The to the emergency department with grow.Pediatrics articles in this supplement provide status epilepticus had systemic examples of some of these varied CSD without adenopathy6 but with published some of the first types of case reports. CSD encephalitis. The authors of reports of the use and effectiveness Downloaded from www.aappublications.org/news by guest on September 30, 2021 S380 MALATACK of propranolol as a treatment of Treatments are offered to improve also are warning case reports.19 One expanding infantile8 hemangioma bronchopulmonary dysplasia10 with relates a vitamin B6 toxicity, and as case reports. These single cases lung reduction surgery, to manage the other relates a drug reaction with did not prove the effectiveness the consequences of an ATP1A2 eosinophilia and systemic symptoms of propranolol itself or, more gene mutation with an N-methyl-D-11 to oxcarbazepine in a 20patient with a importantly, the effectiveness of aspartate receptor antagonist, to Elevatingspecific immunotype Pediatric Practice. propranolol as compared with treat neonatal hyperglycemia12 with the then-standard corticosteroid insulin glargine, and to improve treatment. The finding of efficacy in a intestinal pseudo-obstruction with 13 Finally, case reports that can raise disease that spontaneously regresses pyridostigmine. The authors of the level of practice of the pediatric is always a risky proposition. That 3 other articles suggest a novel community may also be worthy of the apparent clinical response treatment of the erythroderma of 14 publication, but in these instances, (involution of the hemangioma) Omenn Syndrome and discuss vagal – the bar needs to be set high.