Case of Chronic Pancreatic Brucellosis

Angelica C Belo
1
, Eugene W Grabowski
2
, Chi Zhang
3
, David L Longworth
1
, David J Desilets
1
1
Baystate Medical Center, Tufts University School of Medicine. Springfield, MA, USA.
2
General Surgery, South Western Vermont Medical Center. Bennington, VT, USA.
3
North Adams Regional Hospital. North Adams, MA, USA
ABSTRACT
Context Hemosuccus pancreaticus is an
uncommon cause of upper gastrointestinal
bleeding. Chronic infection of the pancreas
with Brucellosis causing hemosuccus
pancreaticus has not been previously
reported.
Case report We describe a case of a 75-year-
old man presenting with a pancreatic mass
and hemosuccus pancreaticus secondary to
chronic pancreatic brucellosis. Polymerase
chain reaction analysis of the pancreatic tissue
was positive for brucella after an initial
positive serology. ERCP revealed bleeding
from the pancreatic duct. Computed
tomography scans of the abdomen
demonstrated an enlarging pancreatic mass.
Endoscopic ultrasound showed a cystic mass
in the body of the pancreas. Fine needle
aspiration revealed granulomata. Selective
mesenteric angiogram failed to reveal the
source of bleeding. The patient eventually
underwent pancreatic resection with
resolution of symptoms.
Conclusion This is the first case of
hemosuccus pancreaticus due to chronic
pancreatic brucellosis reported in medical
literature.
INTRODUCTION
Hemorrhage from the pancreatic duct, termed
hemosuccus pancreaticus, is a rare cause of
gastrointestinal bleeding. It may present as
massive or obscure gastrointestinal bleeding.
In this report, we describe a case of pancreatic
mass secondary to chronic brucellosis causing
hemosuccus pancreaticus in a 75-year-old
man.
CASE REPORT
A 75-year-old male dairy farmer presented
with recurrent hematemesis. He was born and
raised in southern Vermont, where he was a
dairy farmer from childhood on a family
farm. He recalls having brucella-infected
herds in the 1940s and 1950s and drank
unpasteurized milk for many years. He was
referred in June 2005 with a 7-month history
of recurrent idiopathic pancreatitis, anemia,
abdominal pain, chills, sweats and weight
loss. He never smoked tobacco nor drank
alcohol and had no family history of
pancreatitis. He had several upper
gastrointestinal endoscopies at another
hospital which failed to identify a source of
bleeding. An ERCP was performed which
showed active bleeding from the pancreatic
duct consistent with hemosuccus pancreaticus
(Figure 1). There was also a stricture of the main
pancreatic duct at its genu. The biliary ducts

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were normal. CT scan of the abdomen
demonstrated calcification at the head and tail
of the pancreas, an ill-defined hypodense
lesion extending from the superior mesenteric
artery to the pancreatic calcification at the
tail, and two hypodense lesions in the spleen
(Figure 2). Subsequent EUS-guided biopsy of
this pancreatic lesion revealed granulomatous
inflammation. A 5 TU (tuberculin unit)
purified protein derivative (PPD) was
negative as were serologies for Histoplasma
capsulatum, Coccidiodes immitis
and
Blastomyces dermatitidis. Fungal cultures
were obtained because of the granulomatous
histopathology on the initial biopsy. We
cultured for those classes of organisms that
typically produce granulomas. Viral
infections do not typically produce this
histopathology and thus were not sought. An
enzyme immunoassay for Brucella abortus on
serum was positive, but a follow up indirect
fluorescent antibody test against Brucella
abortus was negative. Because of this
discrepancy in serologic findings and the
clinical concern about chronic brucellosis, a
repeat EUS-guided biopsy of the pancreatic
mass was performed. Fresh tissue was
transported same day to the Massachusetts
State Laboratory (Jamaica Plain, MA, USA),
where PCR was positive for Brucella species.
Subsequent cultures of pancreas, blood and
bone marrow failed to grow the organism,
however, and histopathology disclosed no
visible organisms. On the basis of the history,
compatible histopathology and positive PCR,
a diagnosis of pancreatic brucellosis was
made. Doxycycline and rifampin were
initially commenced, but the regimen was
changed to doxycycline plus streptomycin
upon acquisition of the latter drug. After two
weeks of this regimen, he was switched back
to doxycycline plus rifampin. His symptoms
initially improved, but several weeks later the
patient had a further episode of massive
hematemesis associated with abdominal pain.
Examination revealed marked pallor,
tachycardia, and hypotension. His hematocrit
was 27% (reference range: 42-52%) and
serum lipase was 490 IU/L (reference range:
13-60 IU/L). Upper gastrointestinal
endoscopy revealed fresh blood coming from
the ampulla of Vater. Selective arteriography
of the superior mesenteric artery, celiac axis,
splenic artery, and gastroduodenal artery did
not reveal a source of bleeding. Repeat CT
scan of the abdomen showed an increase in
the size of the previously known pancreatic
mass. Because of persistent intermittent
massive bleeding episodes, the patient
underwent subtotal pancreatectomy and
splenectomy with complete resolution of his
symptoms. At operation, a fluctuant 3.0-3.5
Figure 1. Injection of contrast into the major papilla
shows blood gushing out of the minor papilla.
Figure 2. CT scan of the abdomen showing ill-defined
hypodense area in the body and tail of pancreas, also
two hypodense areas inferiorly in the spleen, pancreatic
calcification is present at the tail (arrow).

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431
cm diameter mass was found in the head of
the pancreas. Upon opening the specimen a
large amount of clotted blood extruded from a
dilated pancreatic duct. The mass was an
aneurysmally expanded splenic artery with
internal clot which had a ruptured wall and
blood clot directly communicating with the
pancreatic duct (Figure 3). Histology showed
chronic pancreatitis and aggregates of
lymphocytes. Doxycycline and rifampin were
continued postoperatively for 2 weeks. The
patient is now an insulin-dependent diabetic
and is doing well after one year of follow up.
DISCUSSION
Hemosuccus pancreaticus may pose a
significant diagnostic and therapeutic
dilemma. The bleeding may range from
obscure to massive and life-threatening [1].
Bleeding from the pancreas can occur in the
setting of pseudoaneurysm associated with
acute or chronic pancreatitis [2], pancreatic
pseudocyst, and pancreatic tumor [3]. A
bleeding pseudoaneurysm is a rare but serious
complication with mortality reaching as high
as 40% [4]. The splenic artery is most
commonly involved, followed by the
gastroduodenal, pancreaticoduodenal, and
hepatic arteries [5]. Controversy persists
about the optimal therapy for bleeding
pancreatic pseudoaneurysm associated with
chronic
pancreatitis
(arteriographic
intervention or surgical intervention). Other
uncommon reported causes include primary
splenic artery aneurysms [6] and hepatic
artery aneurysm due to carvenous
hemangioma of the liver [7]. Our patient had
a history of recurrent hematemesis, anemia,
idiopathic pancreatitis, and pancreatic mass
due to brucellosis.
His final episode of bleeding was due to
erosion of a mycotic pseudoaneurysm of the
splenic artery into the pancreatic duct in a
setting of chronic brucella infection and
inflammation.
Chronic pancreatitis secondary to brucellosis
has not been previously described. The
pancreas is a rare target organ for brucellosis.
We identified two cases of acute pancreatitis
secondary to brucellosis in the world’s
literature [8, 9], and no cases of chronic
pancreatitis attributable to this organism.
Although we failed to isolate the organism,
presumably because of antibiotic therapy, the
prior exposure history, positive ELISA,
compatible histopathology of the pancreas,
and positive PCR on pancreatic tissue for the
organism support the diagnosis of brucellosis
as the cause of our patient’s illness.
Brucellosis is an occupational disease
affecting individuals with exposure to the
organism through work with livestock or
animal products. It is characterized by a
variety of often nonspecific clinical symptoms
of fever, myalgia, and general malaise, and
can manifest with localized disorders of
specific organs or tissues [10]. The diagnosis
Figure 3. The pancreas cross sections are of the neck,
body and tail, proceeding from left to right (a.), with
the larger image above showing the body just to the
right of the midline in the patient showing the mycotic
aneurysm of the splenic artery and the fistula into the
pancreatic duct. The neck cross section on the left
shows the “blood-filled” pancreatic duct in the upper
left of the section and the splenic artery to the right.
The body cross section in the middle (also magnified:
b.) shows the actual pathology, with, again the mycotic
aneurysm of the right and the fistula on the left. The
final cross section on the right shows the tail of the
pancreas with the normal splenic artery lower right and
the distal pancreatic duct on the left of the specimen,
empty of blood, on the left.

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JOP. Journal of the Pancreas - http://www.joplink.net - Vol. 8, No. 4 - July 2007. [ISSN 1590-8577]
432
can be established by detection of brucella
species in blood cultures or tissue aspirates.
Like tuberculosis, brucella species can be
notoriously difficult to recover from infected
tissues but available serological tests are
highly specific and sensitive for detection of
brucellosis. The infection is treated with long-
term administration of a combination of
appropriate antibiotics. Therapeutic failure
and relapses, chronic courses and
complications such as osteoarticular disease,
epididymoorchitis, hepatitis, endocarditis, and
neurobrucellosis are characteristic of the
disease [11].
Worldwide, brucellosis remains an important
disease in people and domesticated animals.
This case report further confirms the protean
manifestations of brucellosis and reveals
previously unreported cause of hemosuccus
pancreaticus due to chronic pancreatitis
associated with chronic brucellosis.
Received November 29
th
, 2006 - Accepted
January 31
st
, 2007
Keywords
Brucellosis; Gastrointestinal
Hemorrhage; Pancreas; Pancreatitis, Chronic
Conflict of interest The authors have no
potential conflicts of interest
Correspondence
David J Desilets
Department of Gastroenterology
Baystate Medical Center
Tufts University School of Medicine
Springfield, MA 01199
USA
 
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