CLINICOPATHOLOGIC CONFERENCE*
EDWARD A. GALL, M.D.
From the Departments of Medicine and Surgery, Cincinnati General Hospital,
Cincinnati, Ohio
CLINICAL DATA
History. A 21 year old housewife entered the hospital complaining of pain
in the abdomen. About two months before admission the patient suffered a
sudden attack of sharp midabdominal pain which radiated into the chest and to
the apices of both shoulders. There had been no premonitory symptoms.
During this attack she noted a tender mass to the left of her umbilicus. The
pain was intermittent in character and continued for two days Avith diminishing
severity. At the end of this period both pain and mass disappeared. Thereafter she was able to elicit tenderness on deep pressure but save for some fatigue
remained asymptomatic.
Three days prior to entry, there was an abrupt onset of an identical episode
and again a "lump" was felt in the left side of the abdomen. The pain was
agonizing although intermittent. There was neither nausea nor constipation,
but on the night before admission she vomited precipitously several times. The
vomitus was not blood- or bile-stained. Standing erect caused the pain to
grow worse. At first, lying down produced complete relief, but later this only
caused the knifelike intensity to become dulled, although nonetheless severe.
During the day preceding admission, the mass was noted to increase in size.
Bowel movements were unchanged in frequency or appearance.
The patient had one child, two years of age. The past and family histories
were noncontributory. Menses were regular, the last period having terminated
three weeks before the onset of the present attack.
Physical examination showed a thin, tired-looking young woman, lying quietly
in bed, complaining apprehensively of deep-seated left-sided abdominal pain.
The facies revealed anxiety. There was no icterus. The cardiac findings were
normal; the pulse rate was 60 and the blood pressure 115/65. The lungs were
clear and the respiratory rate was 22.
The abdomen appeared wasted and flabby. At occasional intervals, irregular
peristaltic movements were visible. Just to the left of the umbilicus a lump
was visible and this proved to be a tender, firm, rounded, freely movable mass
measuring approximately 8 x 5 cm. The upper portion of the abdomen was
tender on deep palpation, but there was no rigidity. The liver and spleen were
not enlarged and the extremities and neurologic examination were negative.
The temperature was 98.6 F.
Examination of the blood showed an erythrocyte count of five million with
10.6 Cm. hemoglobin per 100 ml. The leukocytes numbered 26,400 with 90
per cent neutrophils, of which 41 per cent were band forms. The red blood
* Received for publication, December 5, 1947.
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225
cell sedimentation rate (Wintrobe) was 28 mm. per hour. The urine showed
an occasional white blood cell, but was otherwise negative. A Kahn test was
negative.
Plain roentgen films of the abdomen, both in the erect and prone positions,
were negative. Pyelograms after intravenous injection of the dye, were also
negative.
During the initial days in the hospital there Avas little change in the abdominal
findings. The mass remained palpable, exquisitely tender and appeared to
become slightly larger. There were several exacerbations of abdominal pain
° of such intensity as to cause the patient to thrash about in bed. In the main,
however, the distress was dul'. and persistent in character. On the second
hospital day the patient had a chill which was followed by a rise in temperature
to 102.6 F. The fever subsided after twenty-four hours and thereafter only an
occasional rise to 99.5 F. occurred. Urinalysis on the third day showed a trace of
albumin;'the sediment contained 8 to 10 red blood cells and 10 to 12 white blood
cells per high power field. The leukocyte count remained at 26,000 per cu. mm.
Therapy consisted of bed rest, soft and liquid diet, opiates, sulfadiazine and
penicillin. Several soft, partly formed, brown stools were passed spontaneously.
There was no dysuria. On the eighth hospital day, the leukocyte count was
8000 with 63 per cent neutrophils, 29 per cent lymphocytes, 4 per cent eosinophils
and 4 per cent monocytes.
Two days later an operation was performed.
CLINICAL DISCUSSION
Dr. Leon Schiff. "Dr. Felson, would you review the films?"
Dr. Benjamin Felson. "I'm afraid I haven't very much help to offer you.
The plain and upright films of the abdomen show no soft tissue mass. The
colon is fairly well outlined with gas and, as you can see here, there is no displacement. The psoas and kidney outlines are not well shown on these films.
There are no unusual shadows in the hepatic or splenic areas and no calcifications
are seen. In the upright films, the lower portions of the lung fields and the
heart appear normal. In the 'intravenous' pyelograms there is normal excretion
of dye and excellent visualization of both pelves and ureters. No abnormalities
appear on either side."
Dr. Leon Schiff. "The presence of midabdominal pain accompanied by a
mass to the left of the umbilicus suggests the possibility of a pancreatic cyst
secondary to pancreatitis The fact that the abdominal pain and the appearance
of the mass occurred so closely together, however, leads me to exclude a pancreatic cyst of either neoplastic or pseudocysts character as the cause of the
patient's symptoms. A serum amylase might have been helpful, but such a
determination, unfortunately, was not made. The sudden onset of the pain
and its radiation to the apex of both shoulders suggests diaphragmatic irritation
either through the leakage of blood or by reason of the occurrence of secondary
infection.
"The fact that the mass was freelv movable and located to the left of the
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GALL
umbilicus is quite in keeping with a diagnosis of mesenteric cyst. Since mesenteric cysts are rarely pedunculated, pain associated with such a cyst would have
to be explained on the basis of hemorrhage into the cyst rather than by torsion
of its pedicle. The curious radiation of the pain would have to be explained by
leakage of blood from the cyst, although the absence of a more impressive anemia
militates somewhat against such an assumption. The recurrence of the pain
two months after the initial attack and the increase in the size of the mass could
be explained by a second hemorrhage into its substance. The disappearance
of the mass in the interval between attacks could result from the leakage of its
contents into the lesser peritoneal sac, in which case there would be no signs of*
generalized peritonitis. An ovarian cyst Avith a twisted pedicle seems unlikely
in view of the location of the mass, but more particularly because of the radiation
of the pain into the chest. I must admit that I am very unhappy about any
diagnosis I am able to make.
"My impression is that the mass was a mesenteric cyst with recurrent intracystic hemorrhage and leakage into the lesser peritoneal sac."
Dr. B. Felson. "What evidence have you that this was not a pancreatic cyst
with partial evacuation into the lesser sac?"
Dr. Schiff. "This mass became noticeable simultaneously with the pain and
from the description given may even have been present before symptoms directed
attention to it. Cysts of- the pancreas are usually pseudocysts secondary to
some intrapancreatic inflammatory disorder and there is almost invariably an
interval of some days intervening between the abdominal pain and the appearance of the mass. I am inclined to believe this mass antedated the initial episode
of pain, but I feel unhappy about the complete disappearance of the tumor
between the two attacks. Dr. Zinninger, what is your thought about this?"
Dr. M. M. Zinninger. "This case presents a puzzling diagnostic problem to
me, as I imagine it undoubtedly does to others here. The radiation of the pain
in the first attack to the back and tips of both shoulders, suggests that the lesion
was located in the upper part of the abdomen and possibly in the retroperitoneal
region. This may not be significant, however, in view of the fact that subsequently the pain seems to have been localized in the tumor or in the region
of the tumor.
"My first impression on hearing the story of a disappearing painful tumor
was that it might have been a hernia, or a recurrent intussusception but the
absence of signs of intestinal obstruction seems to preclude those lesions. The
fact that the patient was able to eat a soft diet and have normal bowel movements during her ten-day stay in the hospital practically rules out, to my mind,
a lesion of stomach or intestine. The severe, cutting pain may be of help in
diagnosis. Severe pain of this type may occur suddenly in several situations
but chiefly in one of the following three: (1) sudden hemorrhage into a solid
tumor or cyst; (2) sudden distention of a hollow viscus; and (3) ischemia such as
occurs, with volvulus or twist of the pedicle of a cyst or tumor. I t is my belief
that the third of these is accompanied by the most severe pain, such as was
present in this patient, and it is therefore my guess that that was the type of
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lesion present here. Such a lesion might, as in this case, show a disappearing
tumor. If the twist remained, we would expect generalized abdominal pain
from extravasated blood. There would also be leukocytosis without much
fever, due to absorption of degenerative products as the tumor or cyst underwent
ischemic necrosis. Also, as necrosis occurs the pain often becomes less intense.
"Considering the location of the tumor, namely, to the left of the umbilicus,
not many possibilities present themselves. A pancreatic tumor or cyst should
not be so freely movable, nor should it disappear. A mesenteric cyst should
not be painful, nor should it disappear. The torsion of an abnormally mobile
spleen or kidney could give such a syndrome, but the spleen is said to be normal,
and the kidneys normal by intravenous pyelogram. Torsion of the omentum
might be considered, but seems unlikely as it rarely forms a definite mass, never
a disappearing tumor, and usually occurs in fat persons following exercise. It
seems to me that the most likely point of origin of the tumor is the pelvis, for
it is well known that pelvic lesions may be present in the abdomen. We are not
told in the protocol about the findings on pelvic examination, but they may not
have helped in the diagnosis, because frequently tumors of pelvic origin lying
in the abdomen may seem to be entirely free of pelvic connection on bimanual
examination."
Dr. Edward A. Gall. "A pelvic examination was not recorded."
Dr. Zinninger. "That's usually the case, isn't it? At all events, the best
suggestion I can make as to diagnosis is either an ovarian cyst, or a pedunculated
fibroid of the uterus with a twisted pedicle. Several years ago I operated upon
a young woman who had had recurrent attacks of severe left lower abdominal
pain and a disappearing tumor somewhat similar to the mass in this case. Operation disclosed a pedunculated fibroid of the uterus which showed evidence
of having been twisted on its pedicle. I attributed the pain in the shoulders
to bloody extravasation. The transitory hematuria may be due to a degenerated
mass lying on the ureter."
Dr. Gall. "Are there any other comments?"
Dr. David Graller. "I still think this could be a pseudocyst of the pancreas.
But if such a cyst had leaked, there should have been much more evidence of
peritoneal irritation."
Dr. Schiff. "Even if the leak had occurred into the lesser peritoneal sac?"
Dr. Graller. "No, I suppose under such circumstances the evidence of peritonitis would be minimal."
Dr. B. Felson. "I don't think any of the pancreatic cysts we've seen have
been mobile, certainly not as freely movable as this one. On the other hand we
have seen one mesenteric cyst which visualized roentgenographically in the
left paraspinal region, but which disappeared a few days later."
Dr. Zinninger. "I've seen many mesenteric cysts but none with pain. They
disappear sometimes because the patient loses track of them, but I've never
seen one empty spontaneously. They are usually freely movable."
Dr. Carl W. Kumpe. "How do you account for the shoulder pain?"
Dr. Schiff. "I thought that the seepage of blood under the diaphragm could
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explain that. If there had been frank perforation, I should think fever would
have occurred earlier and the abdominal signs would have been much more
generalized."
Dr. Zinninger. "The history cites two identical episodes so that presumably
there was shoulder pain during both attacks."
Dr. Gall. "That is correct."
Dr. Zinninger. "That would certainly lead one to suspect a lesion in the
upper abdomen."
Dr. Schiff. "The mass or something associated with it, such as seepage of
blood could lie in relation to the diaphragm. You do have this sort of referred
pain in association with perforated ulcer, of course."
Dr. Zinninger. "There is no progression of symptoms such as one would
anticipate with the expulsion of irritative material into the free peritoneal cavity.
Both episodes were drastic and there was a high degree of leukocytosis with
relatively little fever. That suggests to me necrosis of tissue due to ischemia.
You get leukocytosis without fever of parallel degree in infarction of a viscus."
Dr. Schiff. "But you may also have that with hemorrhage."
Dr. Gall. "What is your opinion, Dr. Felson?"
Dr. Henry Felson. " I agree with Dr. Zinninger. This strikes me as being
more on the order of an infarction of a cyst or pedunculated tumor than it does
perforation. It would be difficult to fit the two episodes into the diagnosis of
cyst unless one presumed that the cyst refilled and ruptured a second time, or
that the initial symptoms were due to an incomplete rupture followed after two
months by a major perforation. I think with the information available that
that is unlikely."
Dr. MacDonald Wood. "I should like to offer two other possibilities: a congenital reduplication of the colon with volvulus or a diverticulum of the
stomach."
Dr. Kumpe. "Don't you believe that the absence of any significant gastrointestinal symptoms would tend to rule out such diagnoses? I think the history
and findings here are very similar to those which we discussed in another patient
who proved to have a pancreatic pseudocyst due to pancreatitis."
Dr. Gall. "Are there other suggestions?"
PATHOLOGIC
DISCUSSION
Dr. Gall. "I saw this patient clinically and in the face of a rounded, tender,
relatively freely movable mass in the midabdomen, made a diagnosis of twisted
ovarian cyst. The surgeon's pre-operative diagnosis was abdominal tumor,
type and origin unknown.
"The patient was operated on through a left rectus incision directly over the
mass. As soon as the peritoneal cavity was entered, the mass was encountered.
I t was found to be bound by thin, fibrous and fibrinous adhesions to the omentum
which completely encased it. The omentum was partially dissected from it
and the tumor was found to be the spleen, freely movable and ectopic. It was
connected to the upper abdomen by a long pedicle composed in the main of the
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229
splenic vessels. The pedicle was twisted and both the arterial and venous
channels were thrombosed.
"Part of the omentum and the spleen with a short stump of its pedicle were
excised and the abdomen closed without drainage. Postoperatively the patient
continued to have mild pain in both shoulders for several days. This gradually
subsided, however, and she was discharged symptom-free on the tenth postoperative day.
"Microscopic studies showed complete occlusion of hilar vessels by thrombi
which exhibited evidence of early organization. The splenic substance was
massively infarcted except for a thin rind beneath the capsule, the viability of
which was undoubtedly preserved through the medium of the small vessels
evident in the attached adhesive strands which extended to the capsule from
the adherent omentum."
ANATOMIC DIAGNOSES
1. Aberrant spleen with torsion of pedicle,
2. Thrombosis of splenic artery and veins,
3. Massive infarction of spleen.
EDITORIALS
CARDIOLIPIN
Six years ago cardiolipin, as a new phospholipin component of antigen for
serologic tests for syphilis, was announced. Clinicians and serologists welcomed
this announcement for they hoped that this new substance would be the longsought-for key to the simplification or, at least, to the clarification of the serology
of syphilis. They thought that simplification would be achieved by the adoption of a few universally accepted procedures which would utilize cardiolipin.
Thus, the innumerable procedures which used the lipo dal antigens would be
eliminated.
But six years of experience shows that cardiolipin has neither simplified nor
clarified the serology of syphilis. Cardiolipin antigens have been adapted to
the technics which formerly used lipoidal antigens. The number of technical
methods has increased rather than decreased. These new methods are being
used by ever increasing numbers of laboratories.
In the testing of large numbers of specimens from syphilitic individuals, the
performance of cardiolipin antigens has been comparable to that of the older
antigens; but as yet there has not been sufficient testing of material from diverse
nonsyphilitic individuals to warrant any conclusion concerning the specificity
of the modified procedures. Final appraisal of cardiolipin can be achieved only
when it has been applied to a significant volume of authenticated, clinically
diverse testing material. Perhaps this appraisal will be achieved from the
analysis of the mass of data accumulating from the extensive testing now in
progress. If not, then the measure of specific reliability must be determined
by the more cumbersome procedure of original method evaluation studies.
To put it simply, although laboratory observations justify much optimism
concerning the future of cardiolipin in serologic tests for syphilis, it has not been
demonstrated that its use can remove any responsibility from the clinician Avho
interprets the results of laboratory procedures in behalf of the patient for whom
the tests are performed.
Director, Venereal Disease Research Laboratory
J. F. MAHONEY, M.D.
U. S. Public Health Service
Staten Island 4, Areto York
LABORATORY TRAINING FOR RESIDENTS IN THE SPECIALTIES
Certification Boards in the specialties, as a rule, require that six months of
the three year period of graduate institutional training be spent in a hospital
laboratory where the candidates are to receive instruction in the basic medical
sciences. The requirements provide that the instruction be divided into anatomy,
40 per cent; pathology, 40 per cent; physiology and chemistry, 10 per cent; and
bacteriology, 10 per cent. The instructions further suggest that the time assigned to the laboratory may be full time for six months, part time for one year,
or a few hours each week throughout the entire training period. In theory
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