Serous cystic Mimicking Mucinous cystic Neoplasma of Body and Tail Pancreas

Treated with Distal Pancreatectomy with and without Splenic Preserving

Zainul Na’im, Nurhayat Usman, Kiki Lukman

Surgery Departement-Digestive Surgery Subdivision of Dr. Hasan Sadikin General Hospital-Padjadjaran University
School of Medicine-Bandung

Abstract

Back ground: Cystic tumors are the second most common exocrine pancreatic neoplasm,
following only adenocarcinoma of the pancreas in the incidence. The advances in modern
cross-sectional imaging makes the identification of cystic lesion of the pancreas becoming
more common. Surgeon must be familiar with the characteristics and treatment of these
lesions to determine individual management appropriately. Although serous cystic neoplasm
is typically a microcystic lesion, there is a wide range of cyst sizes from micro to macro and
even unilocular cysts have been reported. Thus, the diagnosis is difficult and indications for
surgery are controversial.

Purpose and Objectives: Our case report to present the case of patients with macrocystic
variant of the pancreatic serous cystadenoma that mimicking mucinous type cystic lesion
from imaging modality.

Clinical Case: We present the 2 cases of a 38-year-old and 21-years-old otherwise-healthy

female who was found to have a pancreatic cyst at body and tail of the pancreas. Computed
tomography (CT) revealed a large, well‐defined, enhancing thin‐walled unilocular cystic
lesion17, 53 x10, 94 x15, 93 cm in size arising from body and tail of pancreas (case 1) and
the other showed a homogeneous, 7,7x7,47x7,1 cm sized, unilocular cystic mass in the
pancreatic body that displayed hypoattenuation relative to the adjacent pancreatic
parenchyma and without septation with intra cystic slight enhanced calcification portion
(case 2). The values of serum carcinoembryonic antigen (CEA) and CA 19-9 level was
normal limit. The presumptive diagnosis of a mucinous cystic neoplasm was made and the
lesion was resected by distal pancreatectomy with splenic preserving for the case-2. Surgical
pathology confirmed fibrovascular tissue lined by flat ‘mucine producing epithelium’ without
atypia and with islands of pancreatic parenchymatous tissue and was consistent with a
mucinous benign pancreatic cyst (case 1) and the other case without evidence any mucine
secretion and the lining epithelial representing glycogen within the epithelial cells were
consistent with the findings of the macrocystic form of the serous cystadenoma. Follow-up
postoperative showed no evidence pancreatic leakage both of patients.

Conclusion: Pancreatic macrocystic serous cystadenoma is uncommon and can be

misdiagnosed as a mucinous cystic neoplas. In order to make an accurate diagnosis and to
select appropriate treatment, surgeons must be familiar with these cases.

Keyword : serous cystic, mucinous cystic, pancreatectomy

Introduction

Becourt first described cystic lesions of the pancreas in 1824.(1,2) In 1978, Compagno(1,3) et al
first classified cystic tumors into serous cystic neoplasms (SCNs) and muscinous cystic
neoplasms (MCNs) of the pancreas and identified MCN as a distict disease occurring almost
exclusively in the pancreas body and tail of middle-aged women.(1,2,3,4)

In the 1970s, the clinicopathologic spectrum of mucinous and serous cystic tumors were
described. MCNs span the histologic spectrum from benign to invasive carcinoma. These
tumors contain mucin-producing epithelial and identified histologically by presence of
mucin-rich cells and ovarial like stroma(1,5). Staining for estrogen and progesterone is positive
in most cases(5). The epithelial lining is positive for CKs (CK7, CK8, CK18, CK19), EMA,
and less frequently, CK20, CEA, DUPAN-2 and CA19-9(6,7,8). The immunophenotype of
ovarian-type stroma is similar to the ovarian one with positivity for vimentin, calcretinin,
thyrosine hydroxylase, SMA, α-inhibin, Melan-A, CD99 and Bcl-2(9). Frequently seen in
young women, the mean age at presentation is in the fifth decades. Men are rarely affected.
MCNs are typically found in the body and tail of the pancreas(1,3,5).

The radiologic characteristic of an MCNs on a CT scan is the presence solitary cyst, which
have fine septations and be suronded by rim of calcification. Cross-sectional imaging may not
be able to distinguish between benign and malignant MCNs, however, the presence of
eggshell calcification, larger tumor size, or a mural nodule on cross-sectional imaging is
suggestive of malignancy(3,5).

Compared with MCNs, serous cystic neoplasm (SCNs) have a predilection for the head of the
pancreas and occur in patients with higher median age. Patient commonly present with vague
abdominal pain and less frequently with weight loss and obstructive jaundice. On gross
inspection, SCNs are large and well circumscribed mass. Microscopic examination reveals
multiloculated, glycogen-rich small cysts. Central calcification with radiating septa giving
sunburst appereance, is a radiographic sign on CT in 10% to 20% of patients. Recently,
differential cyst fluid protein expression was observed between SCNs and Intraductal
Papillary Mucinous Neoplasma (IPMNs), with accurate discrimination in 92% of patients.
Although serous cystic neoplasma are generally considered benign, pancreatectomy is
suggested when the diagnosis of malignant disease is uncertain or in symptomatic serous
cystadenomas. Patient with a tumor larger than 4 cm are more likely to be symptomatic and
to display a more rapid median growth rate than patients with smaller than 4 cm. Thus, in
select patients with large (4 cm) or rapidly growing lesions, resection of an SCNs is
appropriate(10).
Case Report 1.

A thirty eight years‐year‐old female patient was admitted to the department of digestive
surgery in Hasan Sadikin General Hospital on January 2018 with the complaint of an
abdominal mass in the left hypochondrium for 3 months. The abdominal mass was initially
localized in the left hypochondrium but gradually increased in size to progress toward the
epigastric region. The mass was not associated with abdominal pain and multiple episodes of
nonbilious, non‐blood‐mixed vomiting. There was no history of fever, jaundice, melena, or
hematemesis. She did not have any history of weight loss or loss of appetite.

Clinical examination revealed slightly distended abdomen with a mass felt in the epigastric
region measuring 15 × 20 cm which was soft, non tender, and not moving with respiration.
There was no shifting dullness or fluid thrill and bowel sounds were heard all over the
abdomen.

Hematological investigations, blood sugar level, renal function tests, liver function tests,
amylase and lipase levels were within normal limits.

A contrast‐enhanced computed tomography (CECT) of the abdomen was planned for further
confirmation of intraabdominal mass. The CECT scan showed a large, well‐defined,
enhancing thin‐walled unilocular cystic lesion 17, 53 x10, 94 x 15, 93 cm in size arising from
body and tail of pancreas. Non enhancement ovoid homogenous cystic lession with septation
areas of calcification observed, which was feature suggestive of mucinous cystic neoplasm of
pancreas. There was no communication of the cystic lesion with main pancreatic duct. Her
blood CEA and CA 19‐9 levels were 5 ng/mL and 33 U/mL, respectively, both of which were
normal range.

A final diagnosis of mucinous cystic neoplasm of pancreas was made, and the patient was
planned for exploratory laparotomy. Operative finding was a huge cyst arising from the body
and tail of the pancreas. The cyst was well defined and pushed the stomach anteriorly and
was adherent to the splenic hilus. The mass located in the lesser sac was mobilized off the
greater omentum and transverse mesocolon, but perforated at the lateral point where dense
adhesions occurred to the splenic hilum releasing small amount of mucinous fluid. The
collapsed cyst was mobilized off posterior gastric wall followed copious irrigation into
abdominal cavity. Distal pancreatectomy was performed in combined antegrade and
retrogrrade fashion. An antegrade approach was performed early in procedure with encircled
the pancreatic neck away from the cystic mass and medial to lateral dissection was proceed.
Retrograde approach was performed because thick adherent of the cystic wall to the splenic
hilum whereby the spleen and pancreas were mobilized lateral to medial en block, thus
providing access to splenic vasculature located superior and behind the pancreas (combined
antegrade and retrograde modular pancreatosplenectomy)(5). The gastrocolic and splenocolic
ligament and short gastric vessel were divided to expose the pancreas and spleen. The inferior
border pancreas was dissected, exposing the retroperitoneal plane behind the gland. The
splenic artery was circumferentially dissected from superior border of the pancreas and
divided. Splenic vein was divided at the confluence with the Superior Mesenteric Vein. The
neck of the pancreas then divided. Spleen, pancreatic tissue and cystic mass were detached
from posterior peritoneal attachment. The pancreatic stump was ligated with non absorbale
material. Postoperative recovery was unremarkable and she was discharged on the seventh
postoperative day. Histology of the excised specimen revealed fibrovascular tissue lined by
flat ‘mucine producing epithelium’ without atypia and with islands of pancreatic
parenchymatous tissue. There was no malignancy seen and was consistent with a
mucinous benign pancreatic cyst. Follow-up after 2 months postoperative showed no
evidence of pancreatic leakage and recurrence.

Case Report 2.

A 21-year-old woman was admitted to our hospital in September 2017 because of an

epigastric abdominal mass that had occurred for the previous 3 months. The patient had no
significant past medical history such as pancreatitis, nor did she have any history of
abdominal trauma. The mass was not associated with abdominal pain and vomiting. There
was no history of fever, jaundice, melena, or hematemesis. She did not have any history of
weight loss or loss of appetite. On the laboratory findings, the results from routine complete
blood count (CBC), serum biochemical studies and urinalysis were all within normal limits;
carcinoembryonic antigen (CEA) and carbohydrate antigen (CA) 19-9 concentrations in the
serum were both within normal limits.

Ultrasonography of the abdomen was performed which showed well‐defined cystic appearing
mass lesion in the left upper quadrant, retroperitoneal in location and that abutted the body
and tail of pancreas supero‐medially. A provisional diagnosis of pancreatic pseudocyst was
made.

Abdominal computed tomography (CT) showed a homogeneous, 7, 7 x7, 47 x7, 1 cm sized,

unilocular cystic mass in the pancreatic body that displayed hypoattenuation relative to the
adjacent pancreatic parenchyma, and this lesion was without septation with intra cystic slight
enhanced calcification portion. The cystic mass was not attached to any of the abdominal
viscera and not had a mass effect on the pancreas, stomach, bowel, aorta, left kidney, spleen,
and splenic vessels. There was no abdominal-free fluid or retroperitoneal lymphadenopathy.
The CT findings were consistent more with a resectable pancreatic cyst.
Laparotomy was performed with midline incision. At exploration a tense cystic mass was
adherent to the greater omentum, transverse mesocolon, and the spleen. The mass located in
the lesser sac was mobilized off the greater omentum and transverse mesocolon. Distal
pancreatectomy was performed in antegrade fashion. After inspection, the neck of the
pancreas was encircled and divided away from the mass with linier stapling device and the
splenic artery and splenic vein were identified at superior border of the pancreas and carefully
preserved. Dissection was proceed medial to lateral fashion. The tail of the pancreas was
mobilized off from the splenic hilum carefully. The pancreatic tissue and cystic mass
completely removed. Histologic examination of specimen without evidence any mucine
secretion. There was no malignancy seen. The lining epithelial representing glycogen within
the epithelial cells were consistent with the findings of the macrocystic form of the serous
cystadenoma. Follow-up after 3 months postoperative showed no evidence of pancreatic
leakage and recurrence.

Discussion.

Cystic neoplasms of the pancreas are rare and comprise 10% to 15% of pancreatic cystic
masses and only 1% of pancreatic cancers(11). They are slow growing indolent tumors with
low-grade malignant potentials and are mostly seen in middle aged women(12). Commonly
asymptomatic, they sometimes reach large sizes prior to diagnosis. Routine use of abdominal
ultrasound, CT and MR has led to an increase in detection of pancreatic cystic lesions and
reduced the average size at diagnosis(13,14). Pancreatic cystic neoplasms consist of Mucinous
cystic neoplasms which commonly arise from the body and tail of the pancreas, Serous
Cystic neoplasms that are almost always benign, Intraductal papillary mucinous tumors
(IPMT) and unusual cystic neoplasms including cystic islet cell tumors. Mucinous cystic
neoplasms include mucinous cystadenomas (65%), proliferative cystic mucinous neoplasms
(30%) and mucinous cystadenocarcinomas (<10%)(15). The macrocystic form of serous
cystadenoma is a uncommon benign neoplasm composed of few, relatively large cysts lined
by uniform, glycogen-rich, cuboidal epithelial cells(16).

Imaging with computed tomography scan (CT) and magnetic resonance imaging (MRI) can
be useful and essential to differentiate pancreatic cystic neoplasm from pseudocyst. Presence
of multiple cysts and internal septations are highly suggestive of tumor(17,18). Besides
imaging, CEA is a highly sensitive tumor marker to differentiate between mucinous and
nonmucinous cyst(17). Cyst fluid amylase level is elevated in approximately 50–75% of the
patients of pancreatic pseudocysts, with pseudocyst being effectively ruled out if amylase
level is below 250 U/L(18). Biopsy of the cyst wall with frozen section examination should
always be performed to differentiate pseudocyst from cystic neoplasm(18,19).

Pancreatic cystic neoplasm sometimes difficult to distinguish from pseudocyst as clinical and
radiological evidences may not be sufficient to make an accurate diagnosis. This may result
in misdiagnosis with inappropriate management. Hence, every effort should be made for their
distinction to avoid internal drainage procedures for neoplasms instead of extirpation(20).

This case report depict similarities of mucinous cystadenoma and the rare form of serous
cystic mass of the body of the pancreas that reside in unusual location. Both of these
morphological entity have similar character from clinical, laboratory and radiological image
based on location, hypoattenuation, septation and intra cystic calcification of the CECT
findings. MCNs are typically found in the body and tail of the pancreas and the usual form of
serous cystadenoma is polycystic which most individual cysts are typically <10 mm with
lesion favour the pancreatic head. The other forms of SCNs are honeycomb (20%) and
oligocystic (macrocystic variant) <10% (cysts can be larger than 20 mm)(21).

According to Kaneto et al, the first description of a macrocystic serous cystadenoma was
made 1992 by Lewandrowski and Warshaw reported rare cases of a variant of serous
cystadenoma composed exclusively of macrocysts that measured up to 8.0 cm in diameter on
macroscopic examination, which showed satellite microcysts during microscopic evaluation.
Based on these findings, the term macrocystic serous cystadenoma was coined to defined a
morphological variant of serous cystadenoma(22,23). Macrocystic serous cystadenoma has been
thought to be the variant of serous cystadenoma, and this can result in diagnostic difficulties
for both the radiologists and pathologists. Several cases that were pathologically confirmed as
‘macrocystic serous cystadenoma’ were preoperatively diagnosed as ‘mucinous cystic
neoplasm’ or as ‘pseudocyst’ by the radiologists and surgeons(24,25,26). The differential
diagnosis includes mucinous cystic neoplasm, pseudocyst, retention cyst, congenital cyst, and
enterogenous cyst. It is very important to differentiate the serous cystadenoma from the
mucinous cystic neoplasm because of the malignant potential of the latter. Microscopically,
the columnar epithelium of the mucinous cystic neoplasm may show cellular atypia, and it
stains for mucins and CEA, while the epithelium of serous cystadenoma does not. In the
absence of inflammatory change and chronic pancreatitis, the pseudocyst and retention cyst
can be excluded in these cases. Therefore, the recognition of such a variant of pancreatic
serous cystadenoma is very important to diagnose and properly manage the patient(27).

Therapeutic management of pancreatic cystic neoplasms has not been standardized and is
continuously evolving. Different factors such as type of neoplasia (mucinous or serous),
patient age, surgical risk, and location and size of the lesion influence decision making(27).
Current consensus recommends resection of all mucinous cystic neoplasms because of their
malignant potential. In this case, with the presumptive diagnosis of a mucinous cystic
neoplasma of the pancreas, surgical resection was decided because of location of the lesion as
well as the age and optimal health status of the patients. Whereas the planned operative
procedure was distal pancreatectomy.

Surgical treatment depends on the location, and extension of the tumor. Distal
pancreatectomy, with spleen preservation, whenever possible, should be performed for cystic
neoplasma of the pancreas located in the body and tail of the pancreas. It is important to
remove the entire cystic tumor, with special care not to rupture the lesion. Spillage of
gelatinous content of the neoplasm may cause tumor implantation and dissemination into the
abdominal cavity, causing pseudomixoma peritoneal. Tumor enucleation should not be
performed due to the elevated risk of complications, mainly pancreatic fistula. In addition,
tumor recurrence may occur at the pancreatic surgical margin(28).

Distal pancreatectomy is a safe procedure in high volume centres. Overall postoperative

morbidity ranging from 5% to 50% and a mortality rate of 0%)(29,30,31,32,33,34) The main
complication, pancreatic fistula, occurs in 15%-20% of cases(35).

The distal pancreatectomy technique was first described in 1913 by Mayo(36) and the spleen-
preserving distal pancreatectomy was outlined in 1943 by Mallet-Guy et al(37). Preservation
of the spleen can be performed with or without preservation of the splenic artery and vein. In
1988, Warshaw described a technique without the preservation of the splenic artery and vein,
ligating the splenic vessels at the hilum(38). Although this method appears technically less
difficult and can be performed in a shorter operating time, it has been associated with a
higher incidence of spleen vascular insufficiency(39). However, this procedure should be
considered in the event of an inflamed or fibrosed splenic artery and vein(38). Spleen-
preserving techniques must be avoided when in the presence of the largest tumors or risk
factors for invasive malignancy, such as the size of the lesion, eggshell calcifications and
mural nodules, in order to perform the complete oncological lymph node disection(40,41,42).
However, these techniques are preferred in all other cases to avoid long term infectious and
haematological complications(38,39,41). Studies comparing patients undergoing distal
pancreatectomy with or without splenectomy show no significant differences compared to
perioperative complications, mean operating time, pancreatic fistula rate, length of hospital
stay and mortality(29,30,31,3541,42).

Conclusion:

Pancreatic macrocystic serous cystadenoma is uncommon and can be misdiagnosed as a

mucinous cystic neoplasm. In order to make an accurate diagnosis and to select appropriate
treatment, surgeons must be familiar with these cases.

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