
S Mirza, SS.Panesar
Corresponding Author: Saqeb Mirza 16 Catherine’s Gate, Haverfordwest, PEMBS, SA61 1NB United Kingdom
Email: saqeb_mirza@yahoo.com
SMJ 2008 53(1): 60
Key
Words: Pancreatic pseudo aneurysm,
Massive Haematemesis, Thrombin Injection
Abbreviations:
CT
Computerised tomography
SMA
Superior mesenteric artery
GDA
Gastroduodenal artery
GIT
Gastrointestinal tract
ERCP
Endoscopic retrograde cholangiopancreatography
EUS
Endoscopic ultrasound guided
Introduction
Pseudo
aneurysms are reported to be a rare1 but potentially lethal
complication associated with chronic pancreatitis. The incidence ranges from 6-9.5% of patients with chronic
pancreatitis2, 3, 4 although in one series a pseudo aneurysm was
found in 17% of all patients operated on for chronic pancreatitis5
thus suggesting a higher incidence. A
very high mortality is reported for this condition6, 7 with some
approaching 90% without some form of therapy8 and ranging from
12.5-40% in patients having some form of intervention.2, 5, 9, 10.
Case
Report
We
present a case of a 31-year-old male admitted to hospital with a history of
three episodes of acute haematemesis and malaena stool.
The haematemesis was massive accompanied by three episodes of black tarry
stool over the preceding four days. His
past medical history included two previous admissions for upper gastrointestinal
bleeding in which no specific source of bleeding was identified on endoscopy and
he was transfused. He had a
fifteen-year history of heavy and regular alcohol intake.
He did not have any previous history of documented acute or chronic
pancreatitis.
Examination
revealed a pale, tachycardic and hypotensive gentleman with a blood pressure of
90/50 mmHg. He did not have any abdominal tenderness. His haemoglobin on admission was 7.2 gm/dl, his clotting
screen, urea and electrolytes and liver function tests were normal and he had an
amylase of 84 iu/l on this admission. Chest
and abdominal x-rays were normal with no calcification visible in the pancreatic
area and no free air under the diaphragm. He
was resuscitated with intravenous fluids and blood and stabilised.
Endoscopy carried out the next day failed to identify any specific source
of bleeding. An abdominal
ultrasound was normal with no features of portal hypertension. CT scan showed atrophy of the body and head of the pancreas
and dilatation of the main pancreatic duct with a large soft tissue mass with a
central area containing contrast intimately related to the third part of the
duodenum and this raised the possibility of active bleeding into a pancreatic
pseudo aneurysm (Figs: 1 and 2).
A nuclear red-cell scan was negative for bleeding into the alimentary
tract. He had been transfused 23
units of blood in total during this admission and persistent intermittent
bleeding necessitated transfer to a specialist centre where a mesenteric
arteriogram demonstrated filling of a false aneurysm from a small posterior
branch of the SMA on selective SMA catheterisation (Fig:
3). Selective coeliac and GDA
angiography showed that these vessels were normal.
Despite several attempts, sub-selective catheterisation of the feeding
vessel to the aneurysm proved impossible and hence constituted a failed attempt
at embolisation. He then underwent an endoscopic ultrasound guided
transduodenal injection of the SMA with thrombin after which he stabilised.
A follow-up CT scan three months later showed that the aneurysm had
disappeared (Fig: 4) and the
patient remains well.
Discussion
Most
reported cases in the literature appear to be associated with chronic
pancreatitis secondary to alcohol abuse. Posttraumatic
pancreatitis11 and cholelithiasis12 however have also been
reported as aetiological factors and duration of pancreatic disease is closely
related to pseudo aneurysm formation, as is splenic vein thrombosis.13
Repeated episodes of pancreatic inflammation or chronic ongoing low-grade
inflammation of the gland and disruption of the duct and ductule system causes
fluid exudation into the surrounding structures and spaces.
The enzymes in this fluid exudate begin to digest and erode the walls of
the adjacent peripancreatic vessels, thus weakening their walls14 and
leading to aneurismal dilatation and necrosis eventually leads to rupture of
this aneurysm. Rupture can occur
into a pre-existing pseudo cyst. Alternatively
necrosis of adjacent structures can cause haemorrhage into the retro peritoneum,
abdominal cavity, gastrointestinal tract or into the pancreatic duct system
itself causing haemosuccus pancreaticus8, 15 or into the biliary tree
causing haemobilia. Erosion of the
adjacent bowel may cause bleeding from the bowel mucosa itself.16, 17
The
splenic artery, the gastroduodenal and the inferior pancreaticoduodenal arteries
are the most frequently involved vessels while the SMA, hepatic, dorsal
pancreatic and gastric arteries are involved less commonly.18
Most
cases of pancreatic pseudo aneurysms are associated with known previous
pancreatitis and a large proportion have pseudo cysts associated with the
condition.2, 5, 9, 10 This
is in contrast to our patient who had no previous documented history of
pancreatitis although his CT scan was suggestive of the disease.
Given
the high associated mortality if not treated in time, a high index of suspicion
is necessary for prompt diagnosis and thence management of pancreatic pseudo
aneurysms. A history of previous
pancreatitis or chronic alcohol abuse with GI bleeding and right upper quadrant
or epigastric pain should prompt the consideration of a pancreatic pseudo
aneurysm in the differential diagnosis19. Previous authors have postulated that rupture into a pseudo
cyst with bleeding into the pancreatic duct and hence into the alimentary system
are probably the most common pattern of bleeding.15
This presents with intermittent bleeding which may be massive and have
devastating consequences. It is
thought that the bleeding is intermittent because increased pancreatic duct
pressure leads to thrombosis of its communication with the pseudo aneurysm and
thereafter the duct decompresses and the clot lyses thus leading to a repetition
of the cycle20. Haemorrhage
may occur over a period of months to years or quite frequently with short
periods between bleeds.11 Haemobilia,
causing biliary colic and jaundice has also been described.21
Less commonly bleeding may occur through the bowel mucosa itself due to
necrosis of bowel wall and fistulation of the aneurysm into the GIT.
Pancreatic duct distension probably causes pain20 which is
characteristically described as crescendo-decrescendo type.22
A sudden fall in haematocrit and an increase in size of a pre-existing
pseudo cyst due to bleeding into it, without overt external bleeding23
and positive guaiac stools with iron deficiency anemia24 are also
occult manifestations of bleeding pancreatic pseudo aneurysms.
Endoscopy
is often negative but helps in excluding other sources of bleeding which may be
important in those with a history of alcohol abuse. These include oesophageal varices and ulcers.
Occasionally bleeding from the mucosa may be visible if the pseudo
aneurysm communicates with the bowel mucosa.
Visualized bleeding from the papilla is reported to be a rare event15
and careful and sometimes prolonged endoscopy with a side–viewing duodenoscope25
may help identify the problem.
Visceral
arteriography currently remains the gold standard according to the literature3,
9, 17, 19, 22 and is especially important in identifying the 20% of pseudo
aneurysms that are too small to be seen by other kinds of imaging.26
It also allows preoperative diagnosis of pseudo aneurysms, their
therapeutic embolisation and serves to identify unusual arterial anatomy27
which may have implications for those requiring major surgical intervention.
Dynamic
bolus CT scanning has become a widely utilised non-invasive modality of
investigation with an 80-100% diagnostic accuracy13, 15 and is also
used to follow up pseudo aneurysms that have been embolised.
Rapid contrast injection is said to be necessary especially if cystic
lesions are identified in the pancreas or peripancreatic area12 as it
is important to distinguish between pseudo cysts and pseudo aneurysms that
contain thrombus. This is because
an attempt at drainage of a lesion suspected to be a pseudo cyst that eventually
turns out to be a pseudo aneurysm could be devastating.28
Thus this technique is helpful in identifying pseudo aneurysms in
asymptomatic patients who have no evidence of bleeding.12
Ultrasound
is a less well-evaluated imaging modality for pseudo aneurysms.
Doppler ultrasound identifies turbulent arterial flow within a peri-pancreatic
swelling, thus confirming a pseudoaneurysm29 while simultaneously
observing neighbouring vessels30 and in some centres is used as a
monitoring tool for successful embolisation.31
ERCP
may demonstrate disruption in the ductal system and can be used to define the
anatomy of the duct system, may be used therapeutically in pseudo cyst treatment
and may also identify a clot in the pancreatic duct confirming the diagnosis of
a haemosuccus pancreaticus.11
The
guidelines for the treatment of pancreatic pseudo aneurysms are not well defined
probably because of the rarity of the condition and the lack of enough material
for large studies. Surgical
treatment is a well established form of treatment and has been the traditional
treatment up until the era of endoscopic and endovascular techniques.
It is reported that 7-17% of patients with the condition will require
operative intervention.5, 16 These
include people in whom other modalities of treatment have failed and those who
are good operative candidates where it offers a definitive treatment for the
cause of the pseudo cyst and pseudo aneurysm and also include patients who are
haemodynamically unstable and require exploration.
But surgical therapy is not without its complications with a mortality
ranging from 20-30%9, 16 with a higher mortality if resection
involves the head of the pancreas.11, 13
Most frequently performed, are operations that resect the pseudo aneurysm
with the involved pancreatic area.11, 15, 18, 32
Some studies show results similar to radical surgery by proximal and
distal ligation of the pseudo aneurysms with internal drainage.5, 16
Visceral
artery embolization is considered by some to be the first line treatment for
bleeding pseudoaneurysms17, 26, 33, 34 and has been used alone for
the definitive treatment of uncomplicated pseudoaneurysms.24, 29,35
This is done most often with metallic coils on either side of the
arterial defect36 and success rates of 79% have been reported with
this treatment.37 This is also used as an emergency measure to stop massive
bleeding and buy time so that elective definitive surgery under controlled
conditions can be planned. However,
as in our patient, it is not always possible to sub selectively catheterise the
vessel concerned.
Ultrasound
guided injection of thrombin is a well-recognised technique for the treatment of
peripheral pseudoaneurysms.38, 39, 40 However to our knowledge there are only two previous cases in
which thrombin has been used for pancreatic pseudo aneurysms and in both these
cases ultrasound guidance was external, with thrombin injection being
percutaneous.41, 42 One
of the complications of this technique is that recanalisation may occur and
hence this method requires regular subsequent surveillance.42
Our
case demonstrates that EUS guided thrombin injection can be used successfully to
treat pancreatic pseudo aneurysms especially if endovascular embolisation has
failed. We suggest that this method
can also be used as a method of controlling haemorrhage from a pseudo aneurysm
and thus conversion of a potential emergency operation with an associated high
mortality, to an elective procedure under controlled conditions thereby
potentially reducing the patient’s risk of mortality. There is not enough data to indicate that this method can be
used as definitive treatment for bleeding pseudo aneurysms and regular follow-up
is needed for these patients.
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