his quote by Sir William
Osler, the father of Modern Medicine, is a testimony to the continual
teaching value of case reports. Case reports are a description of a
single case or cases with unique features experienced by physicians,
which are then shared with the scientific community [2]. These are the
first line of evidence in health care system, and are important in
communicating something new learnt from clinical practice [3].
Why Write a Case Report?
We all know that case reports are ranked quite low in
the hierarchy of evidence-based medicine [4], but still retain value in
scientific publishing as an important mode of providing new knowledge to
the field of medicine. They provide a great opportunity for young
researchers to develop their writing skills, perform literature search,
experience the peer-review process, and start their scientific writing
careers [5,6]. They are instrumental in formulating novel ideas that
trigger clinical trials for future research. Albrecht, et al. [7]
analyzed all the case reports and case series published in the Lancet
from January 1996 to June 1997, and found 23.3% (24/103) to be followed
by randomized controlled trials on the same topic. Case reports are the
only medium for reporting unique cases, rare associations, atypical
presentations, and unexpected outcomes. Reporting adverse drug reactions
by means of case reports are fundamental part of pharmacovigilance [8].
Both the first heart transplant by Christian Bernard in 1967, and the
first ever report of people with Acquired Immunodeficiency Syndrome (not
recognized as such yet) in 1981 were published as case reports [9,10].
The various situations where one may think of publishing a case report
are illustrated in Box I.
BOX
I Common Clinical Experiences Which may be Shared by Case
Reports |
• To report a rare or unknown
disorder
• To describe an atypical
etiology or presentation
• To discuss a challenging
differential diagnosis
• Rare
manifestations/complications of a known disease
• Rare associations with
sound justification
• New insights into
pathogenesis of a disease
• To report unusual
drug-interactions
• To describe new/rare
side-effects of drugs
• To prompt or disconfirm a
hypothesis
• To report any novel
diagnostic procedure
• To report a new treatment
modality
• To report an unexpected
outcome
• Common topics with high clinical relevance
|
Structure of a Case Report
A case report should be a structured, brief and
focused document highlighting a clear learning-point. Case reports do
not follow the IMRAD (Introduction/Methodology/Results/Discussion)
structure that is followed in research articles, but still follow the
underlying schema of Why (you are reporting the case?), How
(it was done?), What (was found?), and What (it all
means?). There is minor variability amongst journals, but mostly the
following sections are present in a case report:
Title: The title of the case report is obviously
one of the first thing seen by the reader, reviewer or editor. The title
should give them an idea regarding what is being reported. The title
must not describe all the findings, but create a sense of curiosity
amongst the readers. Therefore, it should be simple, specific, concise,
catchy and eloquent [11,12]. Some of the journals, and the
Consensus-based Clinical Guidelines Case-Reporting (CARE) [13], suggest
that the title contains the words ‘case report’, e.g.,
‘Headache and transient visual loss as the only presenting symptoms of
vertebral artery dissection: a case report [14].’
Usually, it is preferable to write the title after
writing the whole manuscript, as it is at that time that one has a
better overall impression of the key message you wish to convey.
Abstract: It is a brief and short description
(50-100 words) emphasizing the prudent points of the case, and anything
new added to the existing knowledge. Abstracts for case reports are
generally unstructured, and some journals might not even ask for an
abstract. However, a 4-point structured abstract is required by
Indian Pediatrics, developed after discussions among a variety of
stakeholders [15]. The CARE guidelines also have recommended a
structured abstract.
Introduction: The introduction should be short,
and describe the background regarding the case to be reported, and what
is already known. A justification regarding why we need to report this
has to be given with what has already been reported in the past and what
new will it add to the existing knowledge. Some journals do not even ask
for an introduction and the manuscript starts with the description of
the case [16].
Case-description: The description of the case
must report all events in a chronological order. This section describes
the history, clinical examination findings, demographic data and all
investigation reports that support the diagnosis, and exclude the other
differential diagnosis. The treatment and follow-up must be described in
detail, and all important negative findings must also be included. The
case-description must be sufficiently detailed in order to give an
opportunity to the readers to make their independent clinical impression
and differential diagnoses. This may be supported with figures like
clinical photographs, radiological images, and photographs of
pathological specimens/slides. All information conveying the case-detail
must be included, while avoiding superfluous details that may break the
flow [12,16].
Take care not to detail all the investigations
carried out; only the relevant ones need to be detailed with actual
values (preferably with normal range, if it is an uncommon or
specialized investigations and majority of readers are unlikely to be
aware of the normal values) and units as per the journal’s requirements.
Rest of the investigations carried out can be clubbed together and
stated to be ‘within normal limits’ or by some such statement. Some
journals may even allow you to add a table to detail all the
investigations, if the work-up was too exhaustive, or the patient was
followed-up for a long period; refer to past issues or the authors’
instructions of the concerned journal for guidance.
It is preferable to provide the relevant
imaging/micrograph pictures with the submission; these may or may not be
included in the final publication but allow the reviewer to make an
informed decision about your work-up and diagnosis. Always take a
written, informed consent from the patient/parents/legal representative
for the publication of the case report, including photographs and
clinical details (after showing them to the patient/parents). This
precludes problems later, when the patient is non-contactable after
discharge, and the journal asks for a permission letter.
Discussion: This is a crucial part of the
manuscript, and justifies why the case is worth reporting. This section
should start with a summary of the salient features of the case,
followed by comparison with similar cases reported in the past and
reasons why the presented case is different. Do not claim ‘first such
case’ as your literature search strategy may not be systematic or
comprehensive, and is likely to miss similar cases reported in
literature. The discussion evaluates the case for its novelty,
uniqueness, variability and appropriateness, comparing with literature
published in the past to derive any new knowledge and applicability in
clinical practice [2]. The justification for the present
diagnosis/intervention must be discussed, followed by some implications
of the case on clinical practice. As case reports have a low level of
evidence, we must not make overambitious generalized recommendations;
rather, limit ourselves to enlist the learning points that add to
existing knowledge, and make some appropriate and specific suggestions
depending on the quantity of literature available [17].
References: The number of references permitted
for a case report is very limited; this varies amongst journals –
Indian Pediatrics permits a maximum of 10 references. You do not
need to cite all previous such cases published, but the more relevant
and preferably the recent ones.
Authorship Issues
Most editors will agree that authorship issues are
most commonly encountered with case reports. Both gift-authorship and
exclusion of deserving authors (ghost authors) is common. This has led
quite a few journals (e.g., Indian Pediatrics, BMJ Case
Reports) to limit the number of authors permitted for case reports.
These authorship issues quite frequently arise as the
authorship criteria provided by various groups like the ICMJE are not
easily applicable to case reports. Moreover, as it is not a pre-planned
‘study’, very frequently the requirement ‘substantial contributions to
the conception or design of the work’ is missing, and it is also
difficult to differentiate ‘acquisition, analysis, or interpretation of
data’ from routine clinical care. Though, the remaining three criteria
remain valid. Thus, it has been suggested that one authorship criteria
could be "all authors must have made an individual contribution to the
writing of the article and not just been involved with the patient’s
care" [18].
Another needling issue is the authorship opportunity
to members of investigative or supportive departments e.g.,
Radiodiagnosis, Pathology, Pediatric surgery. As the admitting
department/unit has the access to the patient data and follow-up,
supportive departments may frequently miss on the opportunity to report
the case; even though it may have been their contribution that led to
the diagnosis (imaging or biopsy) or improvement (surgery). There is,
thus, a need for individual institutes to develop guidelines or standard
operating procedures for reporting of cases and the ownership of the
data. A time-limit may be set for the admitting department/unit to
prepare and submit the case report, after information to all departments
involved in the care of the patient. An oversight group from these
departments may discuss and decide on the authors.
Till recently, there was a wide variability in
guidelines for writing case reports and instructions to authors of the
specific journals had to be followed. Consensus-based Clinical
Guidelines Case-Reporting (CARE) have recently been proposed by Gagnier,
et al. [13]. The checklist provided [19] is used by many journals
and reviewers while reviewing case reports. These guidelines bring about
completeness in writing the case, thereby increasing their chances of
acceptance.
Limitations of Case Reports
Case reports are known to have some inherent
limitations. Findings are specific to that particular case and cannot be
generalized, and make limited contribution to the scientific
knowledge-base. Moreover, as they are lower in the level of evidence,
they are cited infrequently, very often leading to a detrimental effect
on the journal’s Impact factor. Some journals have limitation of space
and have had incidents of authorship abuse in the past, which has lead
to removal of the case report section from their journal [20,21]. Many
of the interview boards, and even the Medical Council of India [22], do
not count publication of case reports as a ‘research publication’. It is
becoming more and more difficult for authors to get their case reports
accepted in mainstream journals. Current acceptance rate of case reports
by Indian Pediatrics is below 5%. Some common reasons for
rejection of case reports are detailed in Box 2, which may
be helpful to the beginner. However, the concept of journals dedicated
only to publication of case reports is a positive change.
BOX II Common Reasons for Rejection of
Case Reports
|
• Too common condition
• Too rare condition, that
readers are unlikely to encounter (usually indicates a mismatch
with the readership)
• Too obvious diagnosis
• New gene mutation but with
no clinical relevance
• Diagnosis not
robust/required investigations not done
• All differential diagnoses
not ruled out
• Unethical investigation or
treatment
• No teaching point/value
• Unclear message or wrong message
|
Conclusions
Case reports are brief excerpts where clinicians
describe their experience of a particular case. Despite inherent
limitations and limited educational value, they still remain an
important tool for sharing scientific knowledge, and an easy avenue for
polishing the writing-skills of the beginner. The persisting interest of
readers in case and the arrival of many journals primarily dedicated to
publication of case reports, will ensure that this important link in
scientific evidence does not become extinct.
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