Abstract
There is increasing debate about the value of case reports in the medical and psychiatric literature (e.g. [1–4]). On the one hand, case reports may be viewed positively as an expedient, and often the initial, way of communicating new ideas, syndromes, treatments and adverse reactions. They represent a form of publication and study not dependent on large resources or research infrastructure and can be interesting, accessible and readily digestible for readers. The clinical ‘art’ of medicine is readily conveyed through such articles. For authors, they may also provide a means to get started in medical writing. By contrast, case reports constitute the weakest ‘level of evidence’ in medicine or, as one critic bluntly opined, the ‘lowest form of intellectual life’ [2], p.159]. Some of the cited shortcomings are that they are anecdotal, uncontrolled, unrepeatable, unrepresentative, subjectively interpreted, possibly biased towards reporting favourable outcomes, and do not enhance a journal's ‘impact factor’ (a journal ranking based on citations to articles in that journal).
An endangered species
Because of the aforementioned limitations, published case reports are in jeopardy. Some major psychiatric journals have chosen to reduce the frequency with which they publish case reports, to abandon case reports altogether as regular articles, or to relegate them to the Correspondence section [3]. For example, as long ago as 1978, the American Journal of Psychiatry announced that it would no longer publish case reports as full articles [5]. In the British Journal of Psychiatry, 44 (16%) out of 267 articles in 1987 were case reports, compared to only one (0.6%) out of 167 in 1997 [3]. The Editorial Board of the Australian and New Zealand Journal of Psychiatry (ANZJP) has recently reviewed the place of case reports in that Journal [6]; for now, the ‘humble case report’, to borrow the Editor's term, has been afforded a stay of execution, although the criteria for publication have apparently become more stringent [6]. It is anticipated that, as a consequence, the frequency with which case reports will be published in the ANZJP will diminish.
Case reports in the Australian and New Zealand Journal of Psychiatry, 1967–1999
Despite the vehemence with which proponents and opponents of case reports offer their arguments and despite the long history of publication of such articles, there is negligible systematic data about them, either in the psychiatric or general medical literature. We aimed to describe articles containing case descriptions (‘case report articles’) published in the ANZJP. We wished to ascertain the frequency with which these reports have been published, the objective or focus of such articles, the demographic characteristics of patients described, the diagnostic groups represented, the treatments employed, outcome and changes over time. It was hypothesized that case reports would focus on the unusual and would mostly report positive outcomes. It was anticipated that the findings would inform decisions about the place of case report articles in the ANZJP and furnish data about the quality of evidence that these articles provide.
Method
All case report articles published in the ANZJP from the first issue in March 1967 up to and including the December 1999 issue were identified by inspecting consecutive volumes of the Journal in that period. In the absence of an agreed definition, case report articles were defined as those which described in some detail one or several patients. Excluded were republications of earlier work, of which there were two examples [7, 8] and case descriptions in the Correspondence section; in the latter there is often scant information about the patients described.
The authors constructed two instruments, one to examine each article as a whole and the other to examine each case. The following information was obtained from each article: year of publication, number of cases described and the stated or apparent objectives (e.g. to describe an unusual presentation, refute a theory, report an adverse reaction). For each case, these data were extracted: patient's age group and gender, diagnoses, presence of suicidality, presence of comorbid physical illness, treatment details and outcome. The first author read each article and made the various ratings. The majority of survey items required little subjectivity or inference; ratings of diagnosis and outcome were exceptions, but the choice set in each of these items was small and cut-offs were explicit (e.g. in relation to outcome: improvement, no change or deterioration).
Because this is mainly a descriptive study, few statistical tests were conducted. The χ2 test was used to compare groups. Only differences significant at p < 0.05 are reported. Percentages are rounded to the nearest unit. In order to give an indication of changes over time, data are reported in 11-year periods (the time span examined was 33 years).
Results
There were 256 articles describing a total of 479 patients published in the ANZJP from 1967 to 1999. The numbers of articles (Table 1) and cases (Table 2) increased considerably with time, almost doubling every 11 years. The articles represented 13% of all papers (n = 1935) published in the ANZJP in the 33-year period [8]% (1967–1977), [15]% (1978–1988) and 14% (1989–1999).
Descriptive data on the 256 articles describing cases in the Australian and New Zealand Journal of Psychiatry, 1967–1999
Descriptive data on the 479 cases described in ‘Case report‘ articles in the Australian and New Zealand Journal of Psychiatry, 1967–1999
Descriptive data on the articles and the cases are given in Tables 1 and 2. In summary, the majority of articles described one case and, as hypothesized, reported an unusual presentation. The latter is exemplified by titles such as ‘Myeloneuropathy from whipped cream bulbs presenting as conversion disorder’ [9] and ‘Bilateral ulnar nerve paralysis: an unreported complication of drug-induced extrapyramidal rigidity’ [10]. Articles describing a single case were more likely to report an unusual presentation than articles with more than one case (p < 0.001). Most of the cases pertained to adults aged 19–65 years who often had a mood disorder and/or psychosis and not infrequently a comorbid physical illness. Psychotropic medication and psychotherapy were frequent treatments, hospitalization was common and patients usually had a positive outcome.
The most frequent psychotropic medications were antipsychotics, taken by almost one-third of patients (n = 147; 31%), followed by antidepressants (n = 121; 25%), benzodiazepines (n = 59; 12%), lithium (n = 52; 11%), anticonvulsants (n = 30; 6%), and barbiturates (n = 14; 3%). About one-quarter of patients (n = 116; 24%) took more than one class of medication.
The type of psychotherapy was not always specified. Nevertheless, 51 (11%) patients received cognitive–behavioural therapy and 37 (8%) had dynamic psychotherapy. The most common format was individual therapy (n = 151; 32%), followed by couple or family therapy (n = 46; 10%) and group therapy (n = 14; 3%).
To a large extent, published case reports represent the persuasions of editors, referees and authors. Nevertheless, it is of interest that these articles, considered collectively, appeared to reflect many of the broad characteristics of psychiatric patients and the nature of psychiatric practice, as well as some of the changes that have occurred in our discipline over time. For example, the major diagnostic categories and treatments were described through case reports; these articles also captured the recent emphasis on biological treatments.
A way forward
Case reports have been a regular feature of the ANZJP since a paper describing a patient with Folie a Deux [11] was published in the first issue of the Journal. The fact that the profile of these articles has diminished and that there is increasing scrutiny of them lead to the following questions: Is the case report section in the ANZJP worth saving? Can it be saved? What should be the criteria for publication? Should they be studied further? There are also issues relating to patient consent and anonymity which warrant consideration. We address each of these in turn, drawing on our findings where appropriate.
Are case reports worth saving?
It is not inconceivable that at least some of the case report articles published in the ANZJP were considered useful by Journal readers and have made a worthy contribution to the psychiatric literature. As reported in Table 1, many of these articles set out to present new aetiological data, to describe adverse events and to provide information about common clinical conditions, among other objectives. Admittedly, it could not be said that, in the study period, a major syndrome was first described or a revolutionary treatment first unveiled through a case report in the ANZJP. Nevertheless, case reports have that capacity [12]. Further, there were important data disseminated to the ANZJP readership through these articles, such as descriptions of recently recognized clinical entities, such as Lewy body dementia [13], evaluations of new investigative tools, such as Single Photon Emission Computerized Tomography [14], accounts of liver impairment associated with nomifensine [15] and highlighting of the potential occurrence of neuroleptic malignant syndrome with novel antipsychotics [16, 17].
Thus, case reports have their shortcomings but also their virtues. At issue is whether or not they should appear in the ANZJP and represent 13% of published articles. The answer lies partly in the essential role of the Journal which, according to the Editor, is to educate and share knowledge [18, 19]. Assuming that Osler's view, ‘The best teaching of medicine is that taught by the patient himself’ (quoted in [12], p.xi]), has some relevance today, we would suggest that case reports be retained as full articles by the ANZJP, but perhaps account for less than 13% of all papers.
Can case reports be saved?
A stated aim of the ANZJP is to achieve a higher profile in other countries and to be read by a growing international audience [20]. Is this desire for greater influence and status (shared by several Journal contributors [S. Bloch: personal communication]) compatible with retaining the case report section? Indeed, given the tendency of prestigious journals to decrease the frequency with which they publish these articles or to eliminate them, it could be argued that a proxy measure of journal status, alongside manuscript rejection rates [19], has become ‘the absence or infrequency of case reports’.
It has been argued that, for a variety of reasons, the ANZJP has ‘little chance to… wrestle the mantle from the Archives of General Psychiatry’ [21]. It should not try. Instead, it should strive to fashion its own identity and determine its own ‘place in the sun’. If custodians of the Journal keep this in mind, together with the potential positive contributions that case reports can make, then these articles can survive.
Criteria for publication
There is a need for explicit exclusion and inclusion criteria to improve the quality of case reports and guide authors and referees. Cases which do not warrant publication include those that simply report the mundane or expected, no matter how well written. As one correspondent to the British Journal of Psychiatry suggested [22], such case reports are a waste of readers’ time. Similarly, case reports which simply describe exotica and appear to serve no other function should not be published. Articles which, on the surface, may appear humorous but, at another level, may be construed to denigrate or mock the patients described, or those which seem partly designed to titillate the reader, should be rejected. An example from the ANZJP belonging to this category is the report of a middle-aged woman with nocturnal bingeing who awoke one morning with urinary urgency [23], p.320]: She was alarmed to find that she was unable to raise herself from the bed and had the terrifying thought that she had had a stroke… She [eventually] raced naked to the toilet… and [later] discovered the cause of her scrape: her nightgown was glued to the sheets below with 4–5 crushed and melted chocolate Mars bars.
Which case report articles warrant publication? It has recently been stated [3, 24] that case reports should only be published if they suggest hypotheses that can be empirically tested or refute existing hypotheses. This is perhaps too narrow a purview. We would contend that case reports describing an important new syndrome, investigative tool or treatment technique are also valuable. In essence, case report articles that offer something new and lead researchers or clinicians forward are worthy of publication. On the other hand, many case reports describing adverse events can probably be conveyed in a letter to the Editor. This may result in faster publication and more adverse events described, since they will require less work.
Patient permission and confidentiality
Two areas not addressed explicitly by the ANZJP concern patient permission to report the case and preserving patient anonymity. Given the increasing scrutiny of case report articles, the fact that these articles, unlike many other papers, have usually not passed through ethics review committees, and that other journals are adopting positions on these matters, it may be timely for the ANZJP to specify its own requirements and mention these in the ‘Information for Contributors’. Practices vary. The British Journal of Psychiatry requests that patient consent be obtained and submitted with the article [25]. The American Journal of Psychiatry makes no mention of consent issues but asks that ‘careful attention’ be paid to protecting patient anonymity [26]. The British Medical Journal states that consent is generally required, but there may be certain exceptions, for example when the patient was seen a long time ago, or is likely to be dead (or is dead) and there are no living relatives [27, 28]. The British Medical Journal adds that changing patient details to try to disguise the patient is poor scientific practice.
Further research
Case reports have been the subject of almost no research. To our knowledge, this is the first systematic attempt to examine these articles. Further studies are needed. For example, it would seem important to determine the views of readers about case reports (e.g. determining how commonly these articles are read, whether they are found to be useful and, if so, how, e.g. do they alter clinical practice?). In time, it would also be worth ascertaining whether the ‘new breed’ of ANZJP case reports are different from those published previously, and in what ways.
Conclusion
The status of the case report in the psychiatric literature is parlous. As much as these articles are an easy read and often instructive, their ability to maintain a foothold must depend on their capacity to provide information that is new and useful rather than simply odd or entertaining. Caution must be exercised to not use these reports as a vehicle for parading psychiatric exotica, to not overinterpret the generally positive outcomes that are described and to pay due attention to issues of patient consent and anonymity. In our opinion, however, there must always be a place for clinical descriptions that lead to progress. With the above provisos, we believe that the humble case report can still make a useful contribution to the advancement of psychiatry and the education of psychiatrists.