PRACTICAL GASTROENTEROLOGY JANUARY 2010 36

Bleeding Bile (Hemobilia):

An Obscure Cause of
Upper Gastrointestinal Bleeding:
Case Report and Review
of the Literature
A CASE REPORT
Rana Khan
by Rana Khan, Bishnu Sapkota, Grigoriy Goldenberg, Mohamed Mansour,
Vincent Notar-Francesco, Maurice A. Cerulli
Rana Khan, M.D., Fellow Gastroenterology & Hepatology, Department of Medicine/Division
of Gastroenterology and Hepatology, New York Methodist Hospital Brooklyn, New York.
Bishnu Sapkota, M.D., Fellow Gastroenterology & Hepatology, Department of Medicine/Divi-
sion of Gastroenterology and Hepatology, New York Methodist Hospital, Brooklyn, New
York. Grigoriy Goldenberg, M.D., Attending Physician, Department of Medicine, New York
Methodist Hospital, Brooklyn, New York. Mohamed Mansour, M.D., Chief of Gastroenterol-
ogy, Interfaith Medical Center, Brooklyn, New York. Vincent Notar-Francesco, M.D., Chief,
Division of Gastroenterology and Hepatology, New York Methodist Hospital, Brooklyn, New
York. Maurice A. Cerulli, M.D., FACG, ,Director, Division of Gastroenterology and Hepatol-
ogy, Long Island Jewish Medical Center, New Hyde Park, New York and Associate Profes-
sor of Clinical Medicine, Cornell University.
We report the case of a 76-year-old male who developed acute upper gastrointestinal
bleeding (UGIB) and obstructive jaundice secondary to one of the rarer causes of
UGIB, hemobilia (bleeding into the biliary tree). Bleeding was due to pseudoaneurysm
of right hepatic artery after placement of right and left hepatic duct stents. An emer-
gent hepatic angiogram with embolization was performed with successful hemostasis.
Common causes of hemobilia are iatrogenic, trauma, gallstones, inflammation/infec-
tion, vascular disorders and neoplasm. The classic hemobilia triad includes bleeding
INTRODUCTION
U
pper gastrointestinal (UGI) bleeding is associ-
ated with a serious, sometimes grave prognosis
and high cost of care. In the recent study in Spain
(1), UGI bleeding caused six times more hospital
admissions than lower GI bleeding. In a case-series of
1,000 patients (2), causes of UGI bleeding were:
Peptic ulcer disease (PUD)55 percent
Esophagogastric varices14 percent
Arteriovenous malformations6 percent
Mallory-Weiss tears5 percent
Tumors and erosions4 percent each
Dieulafoys lesions1 percent
Others11 percent
Hemobilia is among rare causes of UGI bleeding but
it can be severe and even fatal.
CASE REPORT
A 76-year-old male presented to emergency depart-
ment (ED) with mild, dull mid-epigastria pain and
multiple (45 times per day) episodes of melena
(black, tarry stool) during the last five days. He had
neither hematemesis nor hematochezia. The epigastric
pain was partially relieved by esomeprazole. He also
complained of fatigue, weakness, anorexia, and a ten
pound weight loss over the preceding twelve months.
Past medical history was notable for right nephrec-
tomy secondary to renal cancer, long standing ulcera-
tive colitis status post colectomy with ileoanal
anastomosis, and prostate cancer treated with external
beam radiation therapy.
Two months prior to his acute presentation, the
patient developed painless obstructive jaundice. Sub-
sequent imaging revealed an obstruction at the conflu-
ence of the right and left hepatic ducts, a large stone in
the gallbladder, and a 3.5 cm soft tissue density adja-
cent to the uncinate process of the pancreas (Figure 1).
At a tertiary care hospital, transhepatic metallic stents
were inserted into both hepatic ducts. The etiology of
the intrahepatic lesion could not be verified but was
considered to be most likely malignant (Klatskin
tumor). After stent placement, the jaundice resolved
and the patient felt relatively well.
On admission, the patient was cachectic, had
severe anemia (hemoglobin 5.3 gm/dl) and obstructive
jaundice (total bilirubin 1.9 mg/dl, alkaline phos-
phatase 714 /L, alanine aminotransferase 112 /L,
aspartate aminotransferase 86 /L). His albumin was
low (2.3 gm/dl), Cancer antigen (CA) 19-9 was high
(106.3 /ml) and coagulation profile was normal. He
refused nasogastric lavage, melena changed into volu-
minous hematochezia. On emergent esophagogastro-
duodenoscopy (EGD), profuse bleeding into the
second part of the duodenum originated from the com-
mon bile duct (Figure 2). The patient was bleeding
bile
DISCUSSION
1. Etiology
Hemobilia is defined as bleeding into the biliary tree
from an abnormal communication between a blood
vessel (splanchnic circulation) and bile duct (4,5). The
nosologic term hemobilia was first introduced by
Francis Glisson in 1654, describing a young man dying
from a massive UGI bleed caused by a sword injury to
the liver (3). In modern medicine, Dr. Philip Sandblom
draws attention to hemobilia in Hemorrhage into the
Biliary Tract Following Trauma: Traumatic Hemo-
bilia. (1948) (4) and in Hemobilia (Biliary Tract
Hemorrhage): History, Pathology, Diagnosis and Treat-
ment (1972) (5). In later years, causes of hemobilia
were further analyzed at the case series level.
PRACTICAL GASTROENTEROLOGY JANUARY 2010 37
A CASE REPORT
Bleeding Bile (Hemobilia)
(melena 90%, hematemesis 60%), biliary colic (70%) and obstructive jaundice (60%).
Angiography is the definitive diagnostic and therapeutic procedure with a success rate
of over 90%. In our patient, angiographic embolization provided only temporary rever-
sal of his condition because stents continued to erode into the hepatic arteries. We pre-
sent this case to remind clinicians that hemobilia is a possible cause of UGIB, especially
in patients who present with jaundice, and in patients with biliary stents it can be fatal.
PRACTICAL GASTROENTEROLOGY JANUARY 2010 38
A CASE REPORT
Bleeding Bile (Hemobilia)
Vascular lesions such as aneurysms of hepatic and
cystic artery, vasculitis of cystic artery or portal vein
(1214) tend to cause hemobilia majormassive and
life threatening bleeding (57). Hemobilia associated
with cholelithiasis, acalculous inflammatory conditions
(ascariasis (10), hepatic abscesses (11), cholecystitis/
cholangitis) and neoplasms is usually minor.
Review of case-series (Table 1) demonstrates an
increase in iatrogenic causes of hemobilia during more
recent years. The rate of iatrogenic hemobilia varies
from 1% with liver biopsy (16) or 4% with transhep-
atic cholangiography (17) to 14% with transhepatic
drainage (18) and 40% to 85% during hepatobiliary
surgeries (68). Hemobilia was also reported with
therapeutic anticoagulation (19).
2. Diagnosis
The classic hemobilia triad described by Quinke in
1871 includes UGI bleeding (hematemesis in 60% of
cases and/or melena in 90%), biliary colic in 70% of
cases and obstructive jaundice in 60% of cases (20).
Only 22% of patients present with all three symptoms
(7). Hemobilia can be painless and should be consid-
ered when a patient presents with a GI bleed that is
accompanied by jaundice. Obstructive jaundice can be
caused by an obstructive biliary lesion or by blood
clot. The latter is formed because bile and blood do not
mix. The blood clot may later pass with bile flow or
remain in the duct leading to persistent jaundice. A
prompt EGD is warranted and duodenoscope can be
used to observe bleeding from the ampulla of Vater.
The following features on transabdominal ultrasound
(US) may be suggestive of hemobilia:
A. Blood-containing material in the bile ducts is
echogenic, non-shadowing and can merge with
liver parenchyma.
B. With a clot adhering to the wall of a bile duct, the
size of the duct can be underestimated.
C. Transitions from hyper- to hypoechoic material in
the bile ducts suggest hemobilia.
D. Blood at the walls of the gallbladder can mimic
acute cholesystitis.
A computed tomography CT scan allows imaging
of blood vessels but it is less sensitive in dilatation of
the bile ducts or to detection of intraductal material.
Abdominal angiography is the gold standard for diag-
nosing hemobilia with an approximate 90% likelihood
of finding the bleeding site.
CASE REPORT (Continued)
An emergent hepatic angiogram revealed a large
pseudoaneurysm of the right hepatic artery adjacent to
previously placed metallic stent. Hemostasis was
achieved by successful injection of 5 mm coils into the
(continued on page 40)
Table 1.
% Etiology of hemobilia in case series (19722002).
Trauma Trauma Other/
(accidental) (iatrogenic) Gall Stones Malignancy Inflammation Vascular Unspecified
Sandblom
(n = 355) (5) 1972 38 17 15 6 13 11
Curet
(n = 86) (8) 1984 27 58 15
Yashida
(n = 103) (6) 1987 19 41 9 7 10 14
Green
(n = 222) (7) 2002 5 65 5 7 8 9 1
PRACTICAL GASTROENTEROLOGY JANUARY 2010 40
A CASE REPORT
Bleeding Bile (Hemobilia)
site of arterial erosion (Figure 3). The patients hospi-
tal course was further complicated by cholangitis,
which was conservatively treated with antibiotics.
Obstructive jaundice gradually resolved and the
patient was discharged home
3. Treatment
The main treatment goals are to first achieve hemostasis
followed by removal of the biliary obstruction. As pre-
viously mentioned, the success rate of angiography with
selective embolization is almost 90% (2122). Other
management options include coagulation of the bleed-
ing vessel, surgery or observation. Angiography selec-
tive embolization is associated with less morbidity than
surgical intervention, but still carries a risk of hepatobil-
iary necrosis (6%), abscess formation (9%), bleeding
(6%), and gallbladder fibrosis (2%) (7,8,23,24).
In order to avoid liver ischemia, a potentially fatal
complication, patency of the portal vein must be assured
(continued from page 38)
Figure 1. Imaging. A. Hepatobiliary ultrasound showing marked intrahepatic biliary ductal dilatation (yellow arrows).
B. Abdomen/pelvis showing mass uncinate process of pancreas (grey arrow), S/P right nephrectomy (purple arrow). C. MRI of
abdomen showing mass uncinate process of pancreas (white arrow), gallstone (green arrow), S/P right nephrectomy (purple
arrow). D. MRCP showing stricture at hepatic duct bifurcation (transparent yellow arrow), marked intrahepatic biliary ductal
dilatations (orange arrows) and big gallstone (green arrow).
PRACTICAL GASTROENTEROLOGY JANUARY 2010 41
A CASE REPORT
Bleeding Bile (Hemobilia)
during embolization of hepatic artery. Transient rise in
transaminases usually resolves within six weeks. Sepsis
is a relative contraindication to embolization (24).
Surgery is considered if:
The cause of hemobilia is usually treated surgically,
i.e., cholelithiasis, hemorrhagic cholecystitis, or
respectable neoplasms.
The angiography with embolization fails.
ERCP fails to relieve obstructions.
Surgical hemostasis is achieved by selective ligation
of the bleeding artery or resection of the aneurysm. Sec-
ond-line options include non-selective ligation or liver
resection. Patients with hemobilia minor, for instance
after liver biopsy or cholangiography, can be closely
observed without intervention until cessation of bleeding.
CASE REPORT (Outcome)
Several weeks later the patient was re-admitted with
recurrence of the same disease. He refused angiogram/
embolization and subsequently expired from intract-
able hemorrhage/hemobilia.
4. Mortality
Over the last 30 years, mortality from hemobilia has
decreased from 25% to <5% (7). This decline can be
explained by both a shift toward hemobilia minor due
to an increase in iatrogenic causes (Table 1) along with
the success of angiographic embolization in hemobilia
major (7). Vascular causes of hemobilia carry the high-
est mortality risk.
CASE SUMMARY
The patient presented with the hemobilia triad: GI
bleeding (melena, bloody stool), abdominal pain and
obstructive jaundice. The pain was not a typical colic,
but rather a dull, achy one. These signs and symptoms
along with the pertinent history of biliary tract lesions
and therapeutic interventions was suggestive of hemo-
bilia as the source of the UGI bleed. Hemobilia was
major, profuse, and pointed toward a vascular (arterial)
etiology. In the modern era of stents, arterial erosions
and vascular hemobilia are likely to become more fre-
quent causes of UGI bleed. As depicted by this case,
Figure 2. Esophagrogastroduo-
denoscopy showing: A. Fresh and
clotted blood in the fundus of the
stomach. B. Clotted blood over
the angularis. C. Clotted blood
mixed with bile in the antrum
of the stomach. D. Bulb of the
duodenum showing blood mixed
with bile coming from second
part of the duodenum.
PRACTICAL GASTROENTEROLOGY JANUARY 2010 42
A CASE REPORT
Bleeding Bile (Hemobilia)
this clinical scenario can recur and be fatal. Interven-
tions, such as stent insertions, can have unintended sec-
ondary effects either immediate or delayed as in the
case presented. These untoward secondary effects do
not render the interventions wrong or unethical pro-
vided their initial intent is curative or palliative. n
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Figure 3. Hepatic angiogram. A. Hepatic arteryangiogram showing right hepatic artery (RHA) pseudo aneursym (black arrow).
B. Hepatic artery angiogram showing embolization of pseudo aneurysm (red arrow), right and left hepatic duct stents in
the background (black arrows). C. S/P embolization of pseudo aneurysm (red arrow), hepatic duct stents in the background
(black arrows).