Recurrent Acute Pancreatitis and Wirsungocele

Rajesh Gupta, Sandeep Lakhtakia, Manu Tandan, Darisetty Santosh,
Guduru Venkat Rao, Duvvuru Nageshwar Reddy
Asian Institute of Gastroenterology. Hyderabad, India
ABSTRACT
Context The association of Santorinicele with
pancreas divisum has been well described.
There is an increased risk of recurrent acute
pancreatitis in patients with pancreas divisum
who also have Santorinicele. Focal saccular
dilation of the terminal part of the main
pancreatic duct has been described as an
incidental
finding
and
termed,
‘Wirsungocele’.
Case report We report a case of a 39-year-
old male who had recurrent episodes of acute
pancreatitis. Laboratory tests, US of the
abdomen and CECT of the abdomen
confirmed acute pancreatitis. MRCP showed
focal saccular dilation of the terminal part of
the main pancreatic duct suggestive of
Wirsungocele. An ERCP confirmed MRCP
findings. An endoscopic pancreatic
sphincterotomy was performed and a 5 Fr
single pigtail pancreatic stent was placed. The
pancreatic stent was removed after 4 weeks.
At the 12-week follow-up, the patient was
asymptomatic.
Conclusion This case report describes the
association of Wirsungocele with recurrent
acute pancreatitis.
INTRODUCTION
Cystic dilatation of the intramural portion of
the dorsal pancreatic duct, i.e. Santorinicele,
has been described in association with
pancreas divisum [1, 2]. It has also been
shown that patients with pancreas divisum
who have Santorinicele have an increased risk
of recurrent acute pancreatitis and are more
likely to benefit from pancreatic endotherapy
[3, 4, 5]. Focal saccular dilatation of the
terminal part of the main pancreatic duct has
been aptly termed, Wirsungocele by Baron et
al. [6]. However, it was reported as an
incidental finding. We report a patient who
had recurrent acute pancreatitis and a
Wirsungocele.
CASE REPORT
A 39-year-old male presented to our institute
with a history of recurrent episodes of
pancreatitis during the previous year. His
symptoms had started one year earlier when
the first attack of pancreatitis occurred. The
results of his tests showed elevated serum
lipase (3,046 IU/L; reference range: 5.6-51.3)
and amylase (3,156 IU/L; reference range: 0-
220 IU/L).
Other examinations, which included a
complete blood count, liver function test,
kidney function test, lipid profile and
transabdominal ultrasound, were normal. He
improved with conservative treatment. A
second attack of pancreatitis recurred after 3
months and this also subsided with
conservative treatment. The patient remained
asymptomatic for the next 6 months. In the
past 3 months, the patient had two more

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JOP. Journal of the Pancreas - http://www.joplink.net - Vol. 9, No. 4 - July 2008. [ISSN 1590-8577]
532
episodes of documented pancreatitis which
also subsided with conservative treatment.
Contrast-enhanced CT of the abdomen was
also done which revealed a bulky pancreas.
The patient was referred to our institute for
evaluation and further management. The
examination included a complete blood
picture, liver function tests, kidney function
tests, lipid profile, serum amylase, lipase and
IGg4, and transabdominal ultrasound: all
were normal. An EUS showed a
heterogeneous pancreas. MRCP showed focal
saccular dilatation of the terminal part of the
main pancreatic duct measuring 11x7 mm
suggestive of Wirsungocele (Figure 1). An
ERCP confirmed the MRCP finding (Figure
2). An endoscopic pancreatic sphincterotomy
was performed and a 5 Fr single pigtail
pancreatic stent was placed. The pancreatic
stent was removed after 4 weeks. The patient
was asymptomatic at week 12 of the follow-
up.
DISCUSSION
Santorinicele (a cystic dilatation at the orifice
of the minor papilla) has been well
documented among patients with recurrent
attacks of acute pancreatitis and pancreas
divisum [1, 2, 3]. It has been postulated that
relative stenosis at the minor papilla in
patients with pancreas divisum leads to an
increase in intraductal pressure. This
increased intraductal pressure, in addition to
congenital or acquired weakness in the duct
wall, leads to the formation of a Santorinicele
which, in turn, predisposes further obstruction
at the minor papilla causing recurrent attacks
of pancreatitis [4, 5].
Until now, an association between
Wirsungocele and recurrent acute pancreatitis
has not been reported. Baron et al. described
Wirsungocele as an incidental finding in their
case report. To our knowledge, this is the first
case of recurrent acute pancreatitis in
association with a Wirsungocele. Though
endoscopic US did not show any focal cystic
dilatation of the main pancreatic duct, both
MRCP and ERCP confirmed focal saccular
dilatation of the terminal part of the main
pancreatic duct consistent with a diagnosis of
a Wirsungocele. It is possible that EUS could
not visualize the focal cystic dilatation due to
compression of the duodenal wall by the
echoendoscope. Several theories have been
proposed to explain the etiology and
pathophysiology of terminal cystic ductal
dilatations. It has been postulated that
decreased autonomic innervation of the
sphincter of Oddi leads to non-coordination
Figure 1. MRCP showing focal saccular dilation of the
terminal part of the main pancreatic duct.
Figure 2. Endoscopic retrograde pancreatogram
showing focal cystic dilation of the terminal part of the
main pancreatic duct.

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JOP. J Pancreas (Online) 2008; 9(4):531-533.
JOP. Journal of the Pancreas - http://www.joplink.net - Vol. 9, No. 4 - July 2008. [ISSN 1590-8577]
533
of the sphincter and functional obstruction at
the papillary orifice [7]. The anomalous
pancreatico-biliary junction with a long
common channel (more than 5 mm) has also
been proposed to be an important factor in the
formation of choledochoceles [8]. Some
studies have shown a focal dilation of the
main pancreatic duct in the head region with
increase of age [9].
There was no demonstrable peri-ampullary
diverticulum or anomalous pancreatico-biliary
junction to predispose the ductal wall to
weakness in our patient. However, the
possibility of functional obstruction at the
papillary orifice cannot be ruled out. The
pathophysiological mechanism for Wirsung-
ocele formation is unclear. Whether the
association of recurrent acute pancreatitis and
Wirsungocele is causative or incidental
remains to be established. Similarly, the role
of pancreatic endotherapy is also unsubstant-
iated.
In conclusion, this case report clearly shows
the presence of Wirsungocele in patients with
recurrent acute pancreatitis.
Received April 23
rd
, 2008 - Accepted May
29
th
, 2008
Keywords
Cholangiopancreatography,
Endoscopic Retrograde; Cholangiopancreato-
graphy, Magnetic Resonance
Conflict of interest The authors have no
potential conflicts of interest
Correspondence
Rajesh Gupta
Asian Institute of Gastroenterology
6-3-661, Somajiguda, Hyderabad
500082 India
P
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