NEUROGENESIS
The Case for Case Reports Shamik Bhattacharrya, MD,1 Julie Miller, MD, PhD,1 and Allan H. Ropper, MD2 Case reports have seemingly fallen upon hard times. Once esteemed by William Osler and C. Miller Fisher, these reports are now considered anachronistic. Nonetheless, case reports remain valuable and formed the largest proportion of publications written last year by residents in our training program (the Partners Neurology Residency). Although they are easy to produce, it is reasonable to ask if these modern exercises are of equal significance to the narratives of migraine by John Graham in the 1950s or descriptions of Parkinson disease by James Parkinson in 1817. Even a brief reading of currently published case reports raises doubts about the value of many of them but also emphasizes their utility. We argue here that the case report format remains of considerable merit, especially to the aspiring academic clinician. ANN NEUROL 2014;76:484–486
C
ase reports that add value to the existing literature begin with astute selection of case material, which is a particular problem for residents whose clinical experience spans months or at most a few years. Many cases that are ostensibly exceptional to residents are either chance occurrences of multiple diseases or atypical instances of known disease presentations. For example, in our morning resident conference, we recently discussed a patient with concurrent influenza and herpes simplex virus encephalitis. Several residents wanted to submit this unique combination for publication. The value to the clinical community of publishing this low-probability event, however, is questionable. Similarly, atypical cases such as spontaneous subarachnoid hemorrhage without thunderclap headache or disseminated tuberculosis masquerading as malignancy often lead to discussion among residents about “writing up” the case. If we accept that each person is a unique combination of genetic susceptibilities and environmental exposures, then we should expect that the same disease may manifest differently in each individual. Case reports describing this type of expected variation may not be of lasting worth. By contrast, novel insights into the natural history and treatment of certain diseases are still best reported by single cases and small case series analyzing individual examples. An article published in this journal, for instance, described rabies encephalitis 8 years after expo-
sure.1 This defied previous estimates of rabies incubation time of 1 to 6 months and arguably suggested the existence of a novel, slowly proliferating rabies virus subtype. Likewise, a recent publication described a patient with psychiatric symptoms, low ceruloplasmin levels, and heterozygous pathogenic mutations suggesting Wilson disease in the ATP7B gene, but with no hepatic disease.2 Liver dysfunction had been thought to be a universal finding in Wilson disease. Neither of these articles sought to establish clinical rules from individual cases, but both illustrated our imperfect understanding of the disease, which could prompt further investigation. Case reports are also an important addition to the literature for diseases in which the clinical course, pathophysiology, or management remains unexplored by larger series. An example is metronidazole neurotoxicity, which was described principally by case reports.3 Those articles spurred larger series describing the diverse radiological features and clinical traits of metronidazole neurotoxicity. More recently, anti–N-methyl-D-aspartate receptor encephalitis was initially described in a single case report.4 Many similar neurological diseases or treatments are still unexplored. Although the clinical spectrum of arbovirus encephalitis has been well described, effective treatment, even anecdotal, could be a subject of future case reports, for example. Adverse drug effects undetected during clinical trials are also often first reported in case
View this article online at wileyonlinelibrary.com. DOI: 10.1002/ana.24267 Received Jul 14, 2014, and in revised form Aug 15, 2014. Accepted for publication Aug 22, 2014. Address correspondence to Bhattacharrya, Department of Neurology, Brigham and Women’s Hospital, Boston, USA 02115. E-mail: [email protected] From the 1Department of Neurology, Brigham and Women’s Hospital, and Department of Neurology, Massachusetts General Hospital, Boston, MA; and 2Department of Neurology, Brigham and Women’s Hospital, Boston, MA.
C 2014 American Neurological Association 484 V
Bhattacharrya et al: The Case for Case Reports
reports or small series prior to large-scale postmarketing analyses. Case reports ideally should be starting points for further study on a larger scale, such as interrogation of a database or initiation of a pilot trial for new therapy. This type of work is well within the grasp of residents and junior faculty. Besides contributing to the overall body of knowledge, case reports written during residency add greatly to the educational experience of training. The literature review conducted while writing a case report often forms the basis for future clinical expertise and further academic inquiry. We have seen the emergence of resident experts on Susac syndrome, Erdheim–Chester disease, nonparaneoplastic limbic encephalitis, Wilson disease, and others. These individuals are now consulted by faculty and other residents and serve as resources for the entire medical center. The process of writing itself obliges mental and practical discipline. Although residents compose many hundreds of pages of patient notes, they often do not receive guidance or feedback on the quality of clinical exposition. Describing findings without the shorthand of medical jargon imposes precision in observation. As William Osler commented in 1903, “there is no more difficult art to acquire than the art of observation, and for some men it is quite as difficult to record an observation in brief and plain language.”5 The comment is especially true today, with technological aids to diagnosis and decreased reliance on careful clinical examination. In authoring a publication, residents ultimately learn not only to write but also to think with logic and clarity. In addition to educational value, writing these reports advances resident careers. Publications are markers of interest in academic medicine and provide a measure of accomplishment in a curriculum vitae. An applicant for the post of instructor or assistant professor with multiple published case reports has been more academically productive in comparison to a similar applicant
lacking such publications. This alone certainly motivates many residents to start writing case reports. Before investing hours writing about the next “gee whiz” patient, we recommend the following (Table 1): DO YOUR HOMEWORK. Take time after encountering the patient to consider whether to write about the topic. Many issues that appear exciting at first glance lose luster when re-examined. Search for a larger case series on the same matter. Desist from writing a case report if such a series exists and the patient at hand adds little; there are other forums for educational cases, such as resident sections in journals. Spend time reviewing the literature to conceptualize how the case will advance understanding of the disease. This step will also define the appropriate audience, such as the general physician, neurologist, or subspecialist.
Find a faculty guide who ideally, but not necessarily, has expertise on the topic. Important qualities in a writing mentor are accessibility, timeliness in meeting deadlines, and ability to edit with clarity. A successful mentor can guide the resident through the submission process and teach the skills necessary for clear and concise writing. Many case reports are written in collaboration with other colleagues, especially fellow residents, who have contributed to the intellectual framework of the case report. There may be difficulty excluding additional resident authors who saw the patient at some time, were on a care team, or contributed minimally to the production of an article. Avoid poaching a case if another individual was primarily involved in caring for the patient, but at the same time, gratuitous authorship is bad practice. A faculty mentor can preempt hard feelings among house staff and adjudicate authorship before the project gets too far. The inclusion of neuroradiologists, pathologists, and others not directly clinically involved in the case should be similarly discussed in advance of writing. It is customary for the lead resident to be the first author and the mentor to be the senior author, but other configurations are acceptable. IDENTIFY A MENTOR.
TABLE 1. Sample Case Report Outline
Key Components of a Useful Case Report Title: Indicates the nature of the case and novel finding or insight. Introduction: Describes the current understanding of the disease and the reason the case is instructive. Case Report: Brief synopsis focusing on details most relevant to the disease. Elaborates on the descriptive clinical or scientific aspects that make the case worthy of attention. Discussion: Explains how the case advances understanding of disease. Avoids inflated projections that go beyond the material presented and acknowledges weaknesses in the analysis. Suggests direction for future investigation.
October 2014
485
ANNALS
of Neurology
It is wise to make authorship determinations prior to the initiation of the bulk of the work. IDENTIFY A TARGET JOURNAL. Although there is no certain guide to choosing a journal, it is most useful to read published case reports in various journals and compare them to the proposed submission. Be aware that some open access journals have high rates of acceptance but charge substantial publishing fees, which can exceed $1,000. High-impact journals rarely accept case reports based solely on clinical descriptions, but might find a truly new scientific correlation appealing. The format and length of the case report should, of course, adhere to the guidelines for the journal. Only a few directly relevant references are necessary. For a well-researched topic, 1 to 2 weeks should suffice to write the first draft, which typically undergoes multiple revisions in collaboration with the mentor. REVISE. Take special care while revising to remove
stock phrases of daily clinical use such as “usual state of health” and “of note” along with contractions such as “contrast enhanced.” “On examination” and “presents with” are empty prose; just give the findings. The word “reveal” is an irritant for editors and should be spared for true revelation, not simply a clinical or test abnormality. Also recall that signs are either present (Babinski) or absent (Romberg), and not positive or negative. Overall, the process of writing and revising the manuscript should not stretch out over months and can be accomplished within the constraints of a resident work schedule.
Finally, a resident should not abandon the project after a first rejection. Read the reviewers’ comments carefully and incorporate necessary changes for submission to an alternate journal. Many worthy case reports are never published, but the experience of writing and research and the discipline and expertise gained from producing case reports remain valuable even today.
Authorship S.B. and J.M. contributed equally.
Potential Conflicts of Interest Nothing to report.
References 1.
Boland TA, McGuone D, Jindal J, et al. Phylogenetic and epidemiologic evidence of multiyear incubation in human rabies. Ann Neurol 2014;75:155–160.
2.
Arruda WO, Munhoz RP, de Bem RS, et al. Pathogenic compound heterozygous ATP7B mutations with hypoceruloplasminaemia without clinical features of Wilson’s disease. J Clin Neurosci 2014; 21:335–336.
3.
Giannini AJ. Side effects of metronidazole. Am J Psychiatry 1977; 134:329–330.
4.
Vitaliani R, Mason W, Ances B, et al. Paraneoplastic encephalitis, psychiatric symptoms, and hypoventilation in ovarian teratoma. Ann Neurol 2005;58:594–604.
5.
Osler W. On the educational value of the medical society. Yale Med J 1903;IX:325–336.
Editor’s Note: It may seem cynical for the Editors of Annals of Neurology to publish a NeuroGenesis article on the value
of writing case reports, while systematically turning down the vast majority of such contributions that are submitted to us for publication. Nevertheless, we are partial to case reports (and this article) as a way to encourage scholarship among neurology residents. The advice in this article rings true to me as the Editor-in-Chief; a case report was my first publication in the literature as a newly minted neurologist, based on a patient I had seen as a resident. I vividly remember going through each of the steps that the authors of this NeuroGenesis article describe. In particular, the red pencil editing of my typed manuscript personally by Bud Rowland, who was then the Editor-in-Chief of Neurology, was a priceless component of my education as an author and an editor, as well as a human being. (Bud pointed out, among other things, that I was writing about a 29-year-old woman rather than a 29-year-old female, and he deleted the patient’s race, which was commonly if superfluously used in most clinical descriptions in those days, as irrelevant.) To this day I still encourage residents to “write up” an interesting patient whose story I think has value to other neurologists. The exercise itself has educational value beyond the content of the actual manuscript. On the other hand, I urge prospective authors of case reports to read my recent NeuroGenesis article advising junior neurologists on where to publish their work. As the text above advises, “High-impact journals rarely accept case reports based solely on clinical descriptions, but might find a truly new scientific correlation appealing.” Annals, for example, generally does not publish case reports unless they definitively identify a new mechanism of disease or treatment. However, there are many journals that do publish particularly illustrative cases for their heuristic clinical value, and even if, in the end, the case report goes unpublished (I have a few of those as well. . .), the effort is always educational and an important part of the training of an academic neurologist. — Clifford B. Saper, MD, PhD, Editor-in-Chief
486
Volume 76, No. 4
The Case for Case Reports Shamik Bhattacharrya, MD,1 Julie Miller, MD, PhD,1 and Allan H. Ropper, MD2 Case reports have seemingly fallen upon hard times. Once esteemed by William Osler and C. Miller Fisher, these reports are now considered anachronistic. Nonetheless, case reports remain valuable and formed the largest proportion of publications written last year by residents in our training program (the Partners Neurology Residency). Although they are easy to produce, it is reasonable to ask if these modern exercises are of equal significance to the narratives of migraine by John Graham in the 1950s or descriptions of Parkinson disease by James Parkinson in 1817. Even a brief reading of currently published case reports raises doubts about the value of many of them but also emphasizes their utility. We argue here that the case report format remains of considerable merit, especially to the aspiring academic clinician. ANN NEUROL 2014;76:484–486
C
ase reports that add value to the existing literature begin with astute selection of case material, which is a particular problem for residents whose clinical experience spans months or at most a few years. Many cases that are ostensibly exceptional to residents are either chance occurrences of multiple diseases or atypical instances of known disease presentations. For example, in our morning resident conference, we recently discussed a patient with concurrent influenza and herpes simplex virus encephalitis. Several residents wanted to submit this unique combination for publication. The value to the clinical community of publishing this low-probability event, however, is questionable. Similarly, atypical cases such as spontaneous subarachnoid hemorrhage without thunderclap headache or disseminated tuberculosis masquerading as malignancy often lead to discussion among residents about “writing up” the case. If we accept that each person is a unique combination of genetic susceptibilities and environmental exposures, then we should expect that the same disease may manifest differently in each individual. Case reports describing this type of expected variation may not be of lasting worth. By contrast, novel insights into the natural history and treatment of certain diseases are still best reported by single cases and small case series analyzing individual examples. An article published in this journal, for instance, described rabies encephalitis 8 years after expo-
sure.1 This defied previous estimates of rabies incubation time of 1 to 6 months and arguably suggested the existence of a novel, slowly proliferating rabies virus subtype. Likewise, a recent publication described a patient with psychiatric symptoms, low ceruloplasmin levels, and heterozygous pathogenic mutations suggesting Wilson disease in the ATP7B gene, but with no hepatic disease.2 Liver dysfunction had been thought to be a universal finding in Wilson disease. Neither of these articles sought to establish clinical rules from individual cases, but both illustrated our imperfect understanding of the disease, which could prompt further investigation. Case reports are also an important addition to the literature for diseases in which the clinical course, pathophysiology, or management remains unexplored by larger series. An example is metronidazole neurotoxicity, which was described principally by case reports.3 Those articles spurred larger series describing the diverse radiological features and clinical traits of metronidazole neurotoxicity. More recently, anti–N-methyl-D-aspartate receptor encephalitis was initially described in a single case report.4 Many similar neurological diseases or treatments are still unexplored. Although the clinical spectrum of arbovirus encephalitis has been well described, effective treatment, even anecdotal, could be a subject of future case reports, for example. Adverse drug effects undetected during clinical trials are also often first reported in case
View this article online at wileyonlinelibrary.com. DOI: 10.1002/ana.24267 Received Jul 14, 2014, and in revised form Aug 15, 2014. Accepted for publication Aug 22, 2014. Address correspondence to Bhattacharrya, Department of Neurology, Brigham and Women’s Hospital, Boston, USA 02115. E-mail: [email protected] From the 1Department of Neurology, Brigham and Women’s Hospital, and Department of Neurology, Massachusetts General Hospital, Boston, MA; and 2Department of Neurology, Brigham and Women’s Hospital, Boston, MA.
C 2014 American Neurological Association 484 V
Bhattacharrya et al: The Case for Case Reports
reports or small series prior to large-scale postmarketing analyses. Case reports ideally should be starting points for further study on a larger scale, such as interrogation of a database or initiation of a pilot trial for new therapy. This type of work is well within the grasp of residents and junior faculty. Besides contributing to the overall body of knowledge, case reports written during residency add greatly to the educational experience of training. The literature review conducted while writing a case report often forms the basis for future clinical expertise and further academic inquiry. We have seen the emergence of resident experts on Susac syndrome, Erdheim–Chester disease, nonparaneoplastic limbic encephalitis, Wilson disease, and others. These individuals are now consulted by faculty and other residents and serve as resources for the entire medical center. The process of writing itself obliges mental and practical discipline. Although residents compose many hundreds of pages of patient notes, they often do not receive guidance or feedback on the quality of clinical exposition. Describing findings without the shorthand of medical jargon imposes precision in observation. As William Osler commented in 1903, “there is no more difficult art to acquire than the art of observation, and for some men it is quite as difficult to record an observation in brief and plain language.”5 The comment is especially true today, with technological aids to diagnosis and decreased reliance on careful clinical examination. In authoring a publication, residents ultimately learn not only to write but also to think with logic and clarity. In addition to educational value, writing these reports advances resident careers. Publications are markers of interest in academic medicine and provide a measure of accomplishment in a curriculum vitae. An applicant for the post of instructor or assistant professor with multiple published case reports has been more academically productive in comparison to a similar applicant
lacking such publications. This alone certainly motivates many residents to start writing case reports. Before investing hours writing about the next “gee whiz” patient, we recommend the following (Table 1): DO YOUR HOMEWORK. Take time after encountering the patient to consider whether to write about the topic. Many issues that appear exciting at first glance lose luster when re-examined. Search for a larger case series on the same matter. Desist from writing a case report if such a series exists and the patient at hand adds little; there are other forums for educational cases, such as resident sections in journals. Spend time reviewing the literature to conceptualize how the case will advance understanding of the disease. This step will also define the appropriate audience, such as the general physician, neurologist, or subspecialist.
Find a faculty guide who ideally, but not necessarily, has expertise on the topic. Important qualities in a writing mentor are accessibility, timeliness in meeting deadlines, and ability to edit with clarity. A successful mentor can guide the resident through the submission process and teach the skills necessary for clear and concise writing. Many case reports are written in collaboration with other colleagues, especially fellow residents, who have contributed to the intellectual framework of the case report. There may be difficulty excluding additional resident authors who saw the patient at some time, were on a care team, or contributed minimally to the production of an article. Avoid poaching a case if another individual was primarily involved in caring for the patient, but at the same time, gratuitous authorship is bad practice. A faculty mentor can preempt hard feelings among house staff and adjudicate authorship before the project gets too far. The inclusion of neuroradiologists, pathologists, and others not directly clinically involved in the case should be similarly discussed in advance of writing. It is customary for the lead resident to be the first author and the mentor to be the senior author, but other configurations are acceptable. IDENTIFY A MENTOR.
TABLE 1. Sample Case Report Outline
Key Components of a Useful Case Report Title: Indicates the nature of the case and novel finding or insight. Introduction: Describes the current understanding of the disease and the reason the case is instructive. Case Report: Brief synopsis focusing on details most relevant to the disease. Elaborates on the descriptive clinical or scientific aspects that make the case worthy of attention. Discussion: Explains how the case advances understanding of disease. Avoids inflated projections that go beyond the material presented and acknowledges weaknesses in the analysis. Suggests direction for future investigation.
October 2014
485
ANNALS
of Neurology
It is wise to make authorship determinations prior to the initiation of the bulk of the work. IDENTIFY A TARGET JOURNAL. Although there is no certain guide to choosing a journal, it is most useful to read published case reports in various journals and compare them to the proposed submission. Be aware that some open access journals have high rates of acceptance but charge substantial publishing fees, which can exceed $1,000. High-impact journals rarely accept case reports based solely on clinical descriptions, but might find a truly new scientific correlation appealing. The format and length of the case report should, of course, adhere to the guidelines for the journal. Only a few directly relevant references are necessary. For a well-researched topic, 1 to 2 weeks should suffice to write the first draft, which typically undergoes multiple revisions in collaboration with the mentor. REVISE. Take special care while revising to remove
stock phrases of daily clinical use such as “usual state of health” and “of note” along with contractions such as “contrast enhanced.” “On examination” and “presents with” are empty prose; just give the findings. The word “reveal” is an irritant for editors and should be spared for true revelation, not simply a clinical or test abnormality. Also recall that signs are either present (Babinski) or absent (Romberg), and not positive or negative. Overall, the process of writing and revising the manuscript should not stretch out over months and can be accomplished within the constraints of a resident work schedule.
Finally, a resident should not abandon the project after a first rejection. Read the reviewers’ comments carefully and incorporate necessary changes for submission to an alternate journal. Many worthy case reports are never published, but the experience of writing and research and the discipline and expertise gained from producing case reports remain valuable even today.
Authorship S.B. and J.M. contributed equally.
Potential Conflicts of Interest Nothing to report.
References 1.
Boland TA, McGuone D, Jindal J, et al. Phylogenetic and epidemiologic evidence of multiyear incubation in human rabies. Ann Neurol 2014;75:155–160.
2.
Arruda WO, Munhoz RP, de Bem RS, et al. Pathogenic compound heterozygous ATP7B mutations with hypoceruloplasminaemia without clinical features of Wilson’s disease. J Clin Neurosci 2014; 21:335–336.
3.
Giannini AJ. Side effects of metronidazole. Am J Psychiatry 1977; 134:329–330.
4.
Vitaliani R, Mason W, Ances B, et al. Paraneoplastic encephalitis, psychiatric symptoms, and hypoventilation in ovarian teratoma. Ann Neurol 2005;58:594–604.
5.
Osler W. On the educational value of the medical society. Yale Med J 1903;IX:325–336.
Editor’s Note: It may seem cynical for the Editors of Annals of Neurology to publish a NeuroGenesis article on the value
of writing case reports, while systematically turning down the vast majority of such contributions that are submitted to us for publication. Nevertheless, we are partial to case reports (and this article) as a way to encourage scholarship among neurology residents. The advice in this article rings true to me as the Editor-in-Chief; a case report was my first publication in the literature as a newly minted neurologist, based on a patient I had seen as a resident. I vividly remember going through each of the steps that the authors of this NeuroGenesis article describe. In particular, the red pencil editing of my typed manuscript personally by Bud Rowland, who was then the Editor-in-Chief of Neurology, was a priceless component of my education as an author and an editor, as well as a human being. (Bud pointed out, among other things, that I was writing about a 29-year-old woman rather than a 29-year-old female, and he deleted the patient’s race, which was commonly if superfluously used in most clinical descriptions in those days, as irrelevant.) To this day I still encourage residents to “write up” an interesting patient whose story I think has value to other neurologists. The exercise itself has educational value beyond the content of the actual manuscript. On the other hand, I urge prospective authors of case reports to read my recent NeuroGenesis article advising junior neurologists on where to publish their work. As the text above advises, “High-impact journals rarely accept case reports based solely on clinical descriptions, but might find a truly new scientific correlation appealing.” Annals, for example, generally does not publish case reports unless they definitively identify a new mechanism of disease or treatment. However, there are many journals that do publish particularly illustrative cases for their heuristic clinical value, and even if, in the end, the case report goes unpublished (I have a few of those as well. . .), the effort is always educational and an important part of the training of an academic neurologist. — Clifford B. Saper, MD, PhD, Editor-in-Chief
486
Volume 76, No. 4
