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Cir Ciruj 2010;78:67-72
Solid pseudopapillary tumor of the pancreas: case
report and literature review
José Francisco Camacho-Aguilera,* César Romero-Mejía,** Alfonso Valenzuela-Espinoza***
abstract
Background: Solid pseudopapillary tumor of the pancreas is an epithelial tumor of low malignancy that primarily affects
young women and represents ~1-2% of all pancreatic neoplasms. We present a case of this type of tumor treated in the
General Hospital of Tijuana, Mexico, as well as a review of the literature.
Clinical case: We present the case of a 37-year-old female with symptomatology of early satiety and abdominal disten-
sion. During open cholecystectomy we found a tumor in the body of the pancreas. Biopsy was done, establishing the
diagnosis of solid pseudopapillary tumor of the pancreas. The patient was treated successfully with distal pancreatec-
tomy and splenectomy.
discussion: Solid pseudopapillary tumor of the pancreas is a rare neoplasm. It is more frequent in young women and
has an unknown etiology. Clinical manifestations include abdominal pain, sensation of plenitude or early satiety, abdo-
minal mass, nausea and vomiting. Laboratory tests are usually normal. Computerized axial tomography may show a
large encapsulated heterogeneous mass. Diagnosis is established through biopsy and surgery is the best treatment for
this pathological entity.
Conclusions: One may conclude that the solid pseudopapillary tumor is a differential diagnosis in the presence of pan-
creatic tumors, although due to its rarity it is not the first option to discard. Surgery represents the best treatment for this
pathological entity and should be attempted in all cases, independent of the size of the pancreatic injury.
Key words: solid pseudopapillary of the pancreas, Frantz’s tumor, benign neoplasm of the pancreas.
introduction acinar cell tumor, papillary cystic neoplasm, papillary cys-
tic carcinoma, solid and cystic tumor, low-grade papillary
Solid pseudopapillary tumor (SPT) of the pancreas is a neo- tumor and, finally, Frantz’s tumor.2 It was not until 1996
plasm of uncertain etiology and low frequency. It was first when the World Health Organization assigned it its current
described by Frantz in 1959, principally affecting young name (SPT of the pancreas)3 and then classified it within the
non-Caucasian women and subsequently establishing group of borderline-type tumors of the exocrine pancreas,
the same epithelial origin.1 Throughout its history, it has e.g., with uncertain malignant potential.4
been referred to with several different names including the We present a case of this type of tumor treated at the
mentioned papillary epithelial neoplasm, solid and cystic General Hospital, Tijuana, preparing a brief review of the
literature to discuss clinical aspects, diagnosis, histology,
* Especialista en cirugía general, Querétaro, México prognosis and treatment of SPT of the pancreas. Keeping
** Servicio de Cirugía General, Hospital General de Tijuana, Baja this information in mind, we can establish the suspec-
California, México ted diagnosis in similar cases, which can lead to surgical
*** Servicio de Patología, Hospital General de Tijuana, Baja California,
treatment with fewer postoperative complications and com-
México
plete cure.
Correspondence and reprint requests to Pancreatic cancer is the fourth leading cause of cancer dea-
José Francisco Camacho-Aguilera ths in the U.S. and the sixth leading cause of cancer deaths
Alfonso Reyes 125, Col. El Tintero in Europe. The majority of pancreatic cancers are rare before
76134 Querétaro, Querétaro, México
the third decade life, being more frequent in the seventh and
Tel.: (442) 319 3885
E-mail: scienta_medica@hotmail.com eighth decades.5 The most common malignancy of the pancreas
is ductal adenocarcinoma (85-90%),6 with cystic pancreatic
Received for publication: 3-31-2008 neoplasms less frequent and corresponding to only 1% of pan-
Accepted for publication: 10-8-2009 creatic tumors and 10% of all pancreatic cystic lesions.7
Volume 78, No. 1, January-February 2010 67
Camacho-Aguilera et al.
Clinical Case
We present the case of a 37-year-old female with a history
of two Cesarean deliveries 8 and 17 years previously. No
other significant history was reported. The patient’s pre-
sent condition has a 6-month evolution with progressive
abdominal distention exacerbated by the intake of solids
and accompanied by early satiety. There was no presen-
ce of abdominal pain, sensation of a mass, weight loss or
other significant symptoms. Upon physical examination,
mild abdominal adiposity was demonstrated with present
peristalsis, without pain on palpation and without presence
of masses. The remainder of the examination was normal.
Clinical studies were carried out. Ultrasound reported the
presence of chronic cholelithiasis, the reason for her pre-
vious surgery 4 months previously. Open cholecystectomy figure 1. Double-contrast CT (oral and IV), which revealed a mass
revealed a tumor in the body of the pancreas. We performed involving the body of the pancreas.
an incisional biopsy of the tumor. Histopathology report re-
vealed the presence of SPT of the pancreas.
A CAT scan was subsequently conducted to assess the
extent of the mass, finding a heterogeneous, well-defined
7-cm tumor (Figures 1 and 2). The patient was referred to
our institution for a case study with routine preoperative
examinations and was scheduled for surgery. We performed
exploratory celiotomy with a supraumbilical midline appro-
ach. A solid ~8-cm tumor was found in the pancreatic area
to the left of the superior mesenteric artery. We attempted
to perform a simple enucleation of the mass, but upon ob-
servation of the condition where almost the entire thickness
of the pancreas was affected, it was decided to perform a
distal pancreatectomy with splenic preservation. The proxi-
mal pancreas was closed with vertical mattress sutures with
chromic catgut. Because of hemorrhage originating from
the pancreatic branch of the splenic artery, we decide to per-
figure 2. Additional view of the CT showing the mass with a thin
form a splenectomy with ligation of the short vessels and
capsule and heterogeneous density.
the splenic artery at the site of tumor resection. We placed a
Blake type closed system. Postoperatively, the patient was
kept under epidural analgesia, an antibiotic therapy based gure 4), in addition to areas of old and recent hemorrhage
on ceftriaxone (1 g IV/8 h) and metronidazole (500 mg IV/8 with numerous cholesterol crystal deposits. Nuclear fissure
h) along with total parenteral nutrition (nutritional commer- features were observed in pseudopapillary cells (Figure 5).
cial package of 2000 kcal for 24 h) for 7 days. Good clinical This study was consistent with the diagnosis of SPT of the
outcome was subsequently observed with normal vital sig- pancreas.
ns, limited fluid loss through drainage and adequate oral
tolerance. The patient was discharged from our department discussion
in good general condition.
Histopathological examination revealed the presence of SPT of the pancreas is a rare neoplasm. Since the first des-
a 7 × 6 × 4 cm tumor with a grayish-white color (Figure 3). cription by Frantz in 1959 there had only been slightly more
When dissecting the mass, we found dark brown areas alter- than 400 reported cases in the world literature until 1997,8
nating with microcystic areas and congestive hemorrhagic. up to 452 cases in 2004 in the English literature9 and up
During microscopic analysis, solid areas were noted with to 629 cases in 2006 in 178 studies.10 When searching the
cells arranged around a stroma of fibrocongestive tissue national literature, we were able to find only four reported
with obstructed capillaries (pseudopapillary formations, Fi- cases of SPT of the pancreas.11-13 SPT of the pancreas cons-
68 Cirugía y Cirujanos
Solid-pseudopapillary tumor of the pancreas
titutes of 0.2 to 2.7% of the primary nonendocrine tumors of chyma during early embryogenesis.7,10 We also do not rule
the pancreas2,9,12,14,15 with a predominance in non-Caucasian out certain genetic factors that may favor the origin of SPT
women (>90% of the cases).8 Asian and African women of the pancreas due to its predominance in Asian and Afri-
have been singled out as the most frequent group of patients can women.15
with SPT of the pancreas, especially between the second Furthermore, immunohistochemical analysis can find
and third decades of life16 (average: 24 years, range: 2-72 vimentin-positive, neuron-specific enolase, α-1-antitrypsin,
years).10 The male-female ratio is 1:9.5,17 and males tend to CD10 and CD56 in >90% of the cases.3,16,18 The presence of
have it occur at a more advanced age (average: 31 years).15 β-catenin can reach 100% at the level of the cytoplasm and
In our patient, although female, had an age at the time of 80% in the nucleus in samples of SPT in the pancreas.19 The
presentation slightly more advanced than most of the cases opposite occurs with the presence of synaptophysin, chro-
reported in the world literature. mogranin A, insulin, glucagon, somatostatin, cytokeratin,
The exact cause of SPT of the pancreas is unknown S100 protein, trypsin and chymotrypsin where positivity is
and it is suggested that it may be a tumor derived from the very low.9,12,18 A phenomenon that has been observed with
pancreatic duct cells, acinar cells, endocrine cells,16 pluri- the SPT of the pancreas is the presence of E-cadherin in the
potential pancreatic cells or from the extrapancreatic tissue nucleus, but is completely absent in the cytoplasm and cell
(possibly ovarian) that is attached to the pancreatic paren- membrane.20 At the level of the cell membrane we can find
progesterone receptors in the vast majority of cases.12,16,21
Because of this it is believed that this hormone may play
an important role in tumor growth.15 This belief has been
corroborated in part by observing an increased growth of
SPT of the pancreas during pregnancy.16
At the genetic level the presence of mutations has been
demonstrated in exon-3 of the β-catenin oncogene in 90%
of the samples obtained from SPT of the pancreas from 20
patients, although the presence of nuclear overexpression of
p53 was rare.14 We did not find abnormalities in the ras gene
family, gene/protein p53, p16, K-ras or DCP4.14,22 The pro-
liferative index is <1% in samples of non-malignant SPT of
the pancreas, which increases to 30 to 40% in cases of SPT
of the “malignant” pancreas.23
The clinical picture of SPT includes the presence of
figure 3. Macroscopic image of the resected mass where a capsule abdominal pain (58 to 72% of the cases), although almost
is viewed at the bottom of the pancreas and spleen to the right. 30% of the patients with SPT are asymptomatic and the
diagnosis is established with clinical tests performed for
other reasons.2,15,16,24,25 Less frequent are the presence of an
abdominal mass, jaundice,26 vague abdominal discomfort,16
bloating or early satiety,9,15 nausea and/or vomiting, and
weight loss.25 Given the vague symptoms and the slow
growth of SPT of the pancreas, its detection is delayed. SPT
of the pancreas can reach sizes of up to 20 cm, growing
around the tissues rather than into them, being that they are
relatively soft. For these reasons it is very rare for them
to cause biliary or pancreatic obstruction (even those loca-
ted in the top of the pancreas).27,28 Finally, acute abdomen
due to hemoperitoneum as a result of spontaneous ruptu-
re or intratumoral bleeding is extremely rare in SPT of the
pancreas.15,26,29 In the present case, the patient had vague
symptoms (abdominal distention and early satiety) without
the presence of palpable abdominal mass. Although abdo-
minal pain occurs in most cases of SPT of the pancreas,
figure 4. Microscopic image of tumor showing the presence of in our case the symptom was not observed. However, in
pseudopapillary formations (arrow). studies of these patients, these clinical data have been ob-
Volume 78, No. 1, January-February 2010 69
Camacho-Aguilera et al.
all cases, pancreatic tissue should be preserved as much
as possible. Some surgeons may prefer enucleation only,
while the risk of developing a fistula is high.25 In the case
presented here, we attempted to perform an enucleation at
the beginning of surgery. However, in observing the con-
dition of almost the entire thickness of the pancreas, we
established that there was a high risk for the production of a
fistula. Instead, we decided on a distal pancreatectomy with
a splenectomy in the presence of hemorrhage from certain
branches of the splenic artery. Extensive lymph node dis-
section or resection of adjacent structures were not justified
and, therefore, not performed in our patient.15 The com-
pletion of distal pancreatectomy with splenectomy did not
significantly alter morbidity and mortality when compared
with distal pancreatectomy without splenectomy.34 Tumor
size should not be regarded as a predictor of unresectability
figure 5. Microscopic image showing solid growth and signs of
characteristic nuclear fissures (arrow).
because masses up to 30 cm can be resected without pro-
blem.2 Surgical treatment alone results in patient survival
>90% at 5 years with a recurrence rate of 10 to 15%8,12,23
served relatively often, even without a direct diagnosis of even in the few cases that demonstrated multicentric masses
SPT of the pancreas. Our patient underwent surgery accor- from ectopic pancreatic tissue (in the liver, retroperitoneum,
ding to the diagnosis of chronic cholelithiasis, incidentally mesocolon and omentum)8,10,35 or synchronous metastases.
finding the tumor for which biopsies were done. Incidental Resection of these lesions should be attempted because me-
diagnosis after carrying out a clinical study has been a pat- tastasis is not considered a negative predictor of survival2,29
tern in these patients, although it is unknown how often an (in contrast to an elderly patient who is considered a risk
incidental diagnosis is made during surgery performed for factor for metachronous metastasis).36 Finally, although we
other purposes. Laboratory tests are usually normal and the have studied the CT, we have not yet succeeded in defining
diagnosis of SPT of the pancreas is made through clinical its role in SPT of the pancreas. However, it has been pro-
studies: ultrasound (US), CAT and/or MRI scan. US may posed for use in cases of unresectable SPT or with multiple
show a presence of heterogeneous pancreatic mass and wi- liver metastases but without standardized CT scheme.37
thout septations inside, although in some cases it may be SPT histology revealed a solid, hemorrhagic cystic capsu-
difficult to completely visualize.26,30 Therefore, abdominal lar tumor. Its size commonly reaches 6.5 to 15 cm in diameter,
CAT is a superior study where one can observe a large he- although it can reach larger sizes (up to 30 cm). 16 The exis-
terogeneous mass with presence of a well-defined capsule tence of a capsule and intratumor hemorrhage allows us to
(hyperdense in >70% of the cases). In ~10% of the cases, distinguish SPT from other pancreatic tumors.3,27 During
SPT of the pancreas contains a fluid interior.27 Calcifications microscopic analysis there are solid areas alternating with
are found in 30% of the cases and are generally located at degenerative changes that cause a pseudopapillary pattern
the periphery of the tumor.10,31 MRI also shows a heteroge- characteristic of epithelial cells in several layers around a
neous lesion, which is hyperintense in almost 90% of the central fibrovascular stalk.14,16 Solid areas contain necrosis,
cases.27 In the study by Voss et al. it was suggested that a fluffy macrophages, cholesterol and calcification granu-
fine needle aspiration biopsy be performed guided by an en- les.16
doscopic US, which shows a sensitivity and specificity for In 10 to 15% of the cases of SPT of the pancreas the-
neuroendocrine tumors of 81.6 and 87.5%, respectively.32 re are metastases14,16 or tumor recurrence.13 The sites most
Furthermore, a PET scan can show the presence of an ele- frequently affected by metastases are the liver (28%), vena
vation of 18F-FDG by SPT, although it is a study that may cava (27%) and spleen (17%). Other sites that may have
not add additional information to what was provided by the metastatic masses are peritoneum, duodenum, omentum,
CAT scans or MRIs.33 colon and lung.10 Malignant behavior can be predicted with
The treatment of choice is surgical. Because SPT of the the evidence of perineural or blood vessel invasion, with or
pancreas may be located in the top (33 to 40%), the main without deep invasion of surrounding tissues, 26 high degree
body (14-28%) and bottom (32-50%) of the pancreas, op- of cellular pleomorphism, elevated mitotic index, diffuse
tions for surgical treatment are simple enucleation, distal growth pattern, diffuse growth with extensive tumor necro-
pancreatectomy and pancreaticoduodenectomy.2,9,12,16 In sis and presence of an undifferentiated component (such as
70 Cirugía y Cirujanos
Solid-pseudopapillary tumor of the pancreas
components of sarcomatoid carcinoma).23,38,39 None of these M, et al. Solid-pseudopapillary tumors of the pancreas are geneti-
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ways harbor β-catenin mutations. Am J Pathol 2002;160:1361-1369.
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72 Cirugía y Cirujanos
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