October 26, 2009
Oluwadamilola Olaku MD, MPH.
Describe a case report
Describe the history of case reports.
Recognize potential roles of case reports.
Outline pertinent information for a good case report.
„…Any patient that goes through the door of this
hospital is a potential case report…‟
(The Montreal General Hospital)
Case reports are brief reports describing an isolated
clinical case or a small number of cases. They may describe
new or uncommon diagnoses, unusual outcomes or
prognosis, new or infrequently used therapies and side
effects of therapy not usually discovered in clinical trials.
(Khalid Khan, Peter Thompson)
woman presented with diffuse musculoskeletal pain. At the
age of 58, she had undergone gastric bypass surgery
because of severe obesity. At the age of 59, she underwent
modified radical mastectomy for an estrogen receptor-
positive T1N0M0 right breast carcinoma and received
Initial diagnosis. 16 months after mastectomy, she
presented with low back pain, myalgias, and fatigue.
Alkaline phosphatase was 231 U/L (normal 39-117 U/L).
Bone scan revealed areas of increased activity in the right
hip, right inferior pubic ramus, and multiple left ribs,
interpreted as consistent with metastases.
Mammography of Left breast and CT scans of the chest,
abdomen and pelvis were negative. No biopsies were
During the next several years she continued to experience
waxing and waning diffuse musculoskeletal pain.
CT scans of chest, abdomen, and pelvis were done on two
more occasions , and mammography was done yearly; all
these studies remained negative.
At the age of 64, she consulted another physician with
worsening pain around her rib cage.
She had lost 100 pounds since her gastric bypass surgery,
but she remained severely obese (body mass index,
Vitamin D deficiency was now considered.
Serum calcium was 7.8mg/dL
Alkaline phosphatase 220 U/L (normal less than 130 U/L)
Serum 25-hydroxyvitamin D was undetectable (less than
Parathyroid hormone(PTH) was elevated (338pg/ml; normal
Patient was treated with oral vitamin D (ergocalciferol
50,000 IU weekly for 4 weeks, then monthly) and
At 3 months, her rib pain had resolved. On repeat
laboratory testing, 25-hydroxyvitamin D was 11ng/mL,
calcium 8.5mg/dL, phosphorus 3.5mg/dL, alkaline
phosphatase 184 U/L and PTH 248pg/mL.
Ergocalciferol dose was increased to 50,000 IU twice
Four months later, her 25-hydroxyvitamin D level was still
low (15ng/mL), but she remained pain-free.
Khokhar et al.
There has been a long descriptive history of case reporting
in relation to cancer.
In the book titled Clinical Case Reporting in Evidence-
Based Medicine, Milos Jenicek eloquently describes case
reports as the first line of evidence, where everything
Some of the earliest case reports describing individual
patients afflicted with cancer can be traced back to the
papyrus records of Ancient Egyptian medicine of
approximately 1600 B.C..
These reports were the first recorded incurable tumors of
Case reports describing new
century A.C. (Urteaga et al)
Historically case reports were the subject of
letters between colleagues that ultimately became
the origin of journals.
In January 1832, Thomas Hodgkin reported six
cases to the Medical-Chirurgical Society of
London, two of which were what we know today
as Hodgkin‟s lymphoma. (Altschuler EL)
In 1957, Dennis P. Burkitt described a tumor that
presented as a growth in the angle of the jaw of African
In 1990, Farcet et al. described two patients with a new
type of lymphoma, called Hepatosplenic T-Cell lymphoma
leading to more focused research of this new entity.
More recently researchers have used the case report
format to share unusual treatment combinations or
responses. For example, Treon et al reported in 2004 about
an interesting clinical response to sildenafil in
definitive study may be impossible.
The reporting of similar rare cases, affords the
clinician an opportunity to read this discontinuous
“case series” and inform a rational approach to
A well-written case report or small uncontrolled
series of cases is often the first step in the
discovery of new diseases and
treatments.(Albrecht et al, Ruben RJ)
A good case report has the element of surprise
Impact of local discussions
of distant practices
Published case reports are a wonderful way to experience
the clinical activity of distant practices and should lead to
local discussions, further reading, and thinking with
colleagues. (Bignall J)
Case reports offer an opportunity for young authors to
participate in the preparation of the manuscript.
In 1941, a Johns Hopkins University scholar, Stacy Guild,
PhD, wrote a 231 word histological observation describing
glomus bodies in the human temporal bone.
In the same year, a clinical practitioner in New York, Harry
Rosenwasser MD, operated on a patient with what
appeared to be a carotid body tumor in his ear.
Dr. Rosenwasser communicated with Dr. Guild and
the two gave substance to the final case report
that established the clinical entity we know today
as glomus tumors.
Are Case reports
important in the era of
evidence based medicine?
case reports contain “misleading elements” in clinical
presentation, and that they do “more harm than good” by
emphasizing the bizarre (Hoffman JR).
Clinicians will protest that relegating case reports and case
series to second-class status in the medical literature
means that the babies are being thrown away with the
Evidence-based medicine is exclusively concerned with
finding the best evidence for clinical decisions; for example,
should we apply a particular therapy or a diagnostic test to
a particular patient?
A hierarchy of evidence with the randomized trial “on top”
serves one purpose admirably: the final evaluation of
therapies or tests especially when their clinical value is not
immediately clear cut.
Are case reports necessary in the era
of evidence based medicine?
Case reports and case series, however, have other aims
that are equally important in the progress of medical
science and education.(Vandenbroucke J)
Potential roles of case
reports and case series
Recognition and description of new diseases.
Detection of drug side effects (adverse and beneficial).
Study of mechanisms of disease.
Medical education and audit.
Recognition of rare manifestations of disease.
In the summer of 1999, the use of a case series in the
recognition of a new disease was exemplified by the
epidemic of West Nile encephalitis in New York City.
Case reporting remains important for detection of side
effects of drugs, whether adverse or beneficial, even in our
modern age of “designer drugs” and “pharmacogenomics.”
Case reporting prompts most if not all of a series of
retractions of drugs from the market.
Conversely, a pharmaceutical success story grew from the
detection of unanticipated effects: Sildenafil was developed
from observation of a side effect of an antihypertensive
Use of case reports and case
series. The observation that the nicotine
withdrawal syndrome was linked to depression
paved the way to the use of antidepressant drugs
as smoking cessation agents. (Covey et al)
discovered, thanks to the age-old clinical observations on
disease transmission in families.
A renewed look at a family tree led to the discovery of
maternally inherited diabetes associated with deafness; this
in turn led to further clues to the understanding of
(Ballinger et al,Van den Ouweland et al)
Journal of Medical Case Reports is an open access journal
that is committed to publishing high quality case reports
from anywhere in the world.
For example, by publishing every case report about
spontaneous regression of metastatic renal cell carcinoma
after debulking nephrectomy, a specific search of the
database would help researchers in studying this particular
and interesting phenomena.
(Van den Ouweland et al)
For example, in 1886, Felix Frankel described the first case
report of pheochromocytoma in an 18 year old woman
with bilateral adrenal tumor.
In 2007, almost 121 years later, Neumann et al studied four
living relatives of the same patient reported by Frankel in
1886 and found that the patient had RET mutation and
that her family had multiple endocrine neoplasia (MEN 2).
Meta-analysis or Systemic
reviews of cases and case
one cases of tamoxifen-related ocular toxicity in breast
This review produced a clearer picture of the nature and
distribution of the toxicity, as well as the severity of the
They recognized the difficulty of attributing ocular findings
to tamoxifen and other competing causes of retinal,
macular and corneal abnormalities.
reports and case series reports of primary carcinoma of
the fallopian tube.
This author was able to outline more appropriate patient
Hypothesize on the underdiagnosing of cases
Causes of treatment failure
Lack of controlled trials and usefulness of a second look
laparotomy for monitoring disease response to the
explanatory. Only case characteristics and case outcomes
can be better assessed at this level of clinical research.
Anyone who searches for evidence of what is helpful or
harmful to patients is not satisfied with conclusions drawn
from case reports.
In situations where there is no other more adequate
evidence, the best of the first line of evidence must be
taken into account.
Case reports and the Law
Case reports and the law. Federal regulations
(45CFR46.102(d) and 45CFR164.501) define research as a
systematic investigation, including research development,
testing and evaluation, designed to develop or contribute
to generalizable knowledge.
A case report is a medical/educational activity that does
not meet DHHS definition of research.
Medical education/consultation, such as occurs when a
colleague presents a difficult case or case series at a
teaching conference does not require IRB review
Office of Human Subjects Research (OHSR)
Case report review
Case report review instrument
Impact of case report
on medical research
Impact of case reports on medical research
Impact of case series on medical research
Albrecht et al
How to write a good case
The single message must be clear
Why is observation important?
What does it teach us?
Does it run counter to some cherished truth?
Is it an unexpected association?
Is it a rarity that would otherwise be missed?
Case reports should be reported in an organized and
Case reports are still relevant in present day medicine.
In rare instances where clinical trials are not possible for
ethical reasons, case reports/series might be the only
evidence available to recommend treatment.
Dr. Jeffrey White
Dr. Farrah Zia