Adult pancreatic hemangioma in pregnancy - Concerns and considerations of a rare case

Abstract

Background:
Pancreatic tumors in pregnancy are rare but clinically challenging. Careful diagnostic workup, including appropriate imaging examinations, should be performed to evaluate surgery indications and timing . In the present case a diagnosis of an adult pancreatic hemangioma was made. We were not able to identify a similar case in the very sparse literature on this rare disease.

Case presentation:
A 30-year-old woman at 12 weeks of gestation was diagnosed with a large pancreatic tumor having a cystic pattern based on imaging. Although the preoperative diagnosis was uncertain, patient preference and clinical symptoms and signs suggested surgery. Open distal pancreatic resection including splenectomy was performed, and complete resection of the large cystic tumor was successfully achieved, with no postoperative complications. Although a solid pseudopapillary epithelial neoplasm (SPEN) was suspected, specimen morphology, including immunohistochemistry, supported the diagnosis of an adult benign pancreatic hemangioma.

Conclusion:
Although mucinous cystic neoplasm (MCN) and adenocarcinoma are the most common pancreatic tumors during pregnancy, various other malignant and benign lesions can be encountered. This report adds to the very small number of pancreatic hemangiomas reported in the literature and involves the first patient diagnosed with this rare condition during pregnancy. Careful clinical considerations regarding diagnostic workup and treatments are required to ensure that mother and child receive the best possible care.

Full text

C A S E R E P O R T Open Access
Adult pancreatic hemangioma in pregnancy –
concerns and considerations of a rare case
Jon Arne Søreide
1,4*
, Ole Jakob Greve
2
and Einar Gudlaugsson
3
Abstract
Background: Pancreatic tumors in pregnancy are rare but clinically challenging. Careful diagnostic workup,
including appropriate imaging examinations, should be performed to evaluate surgery indications and timing . In
the present case a diagnosis of an adult pancreatic hemangioma was made. We were not able to identify a similar
case in the very sparse literature on this rare disease.
Case presentation: A 30-year-old woman at 12 weeks of gestation was diagnosed with a large pancreatic tumor
having a cystic pattern based on imaging. Although the preoperative diagnosis was uncertain, patient preference
and clinical symptoms and signs suggested surgery. Open distal pancreatic resection including splenectomy was
performed, and complete resection of the large cystic tumor was successfully achieved, with no postoperative
complications. Although a solid pseudopapillary epithelial neoplasm (SPEN) was suspected, specimen morphology,
including immunohistochemistry, supported the diagnosis of an adult benign pancreatic hemangioma.
Conclusion: Although mucinous cystic neoplasm (MCN) and adenocarcinoma are the most common pancreatic
tumors during pregnancy, various other malignant and benign lesions can be encountered. This report adds to the
very small number of pancreatic hemangiomas reported in the literature and involves the first patient diagnosed
with this rare condition during pregnancy. Careful clinical considerations regarding diagnostic workup and
treatments are required to ensure that mother and child receive the best possible care.
Keywords: Cystic lesion, Hemangioma, Pancreas, Pregnancy
Background
In pregnant women, the acute abdomen is often a chal-
lenge. Careful clinical evaluation, close cooperation be-
tween the surgeon and the gynecologist as needed, and
the application of appropriate diagnostic tools and edu-
cated judgment remain the cornerstones of standard
care [1–6]. Although the diagnostic workup can be
demanding in these cases, a responsible surgeon might
regard appropriate surgical treatment as stressful for the
relatively common diseases encountered during preg-
nancy such as appendicitis [7, 8] and acute cholecystitis
[9, 10]. A surgeon is confronted by even greater responsi-
bility for a mother and her child in clinical settings involv-
ing rare acute abdominal conditions of pregnancy, which
include acute severe pancreatitis [11], gastrointestinal
bleeding [12, 13], and suspected tumors in various loca-
tions [14–19]. In cases involving a cystic lesion, the nature
of the lesion, including its malignant potential and the risk
of a spontaneous rupture, must be considered when dis-
cussing indications for and the timing of surgery [15, 20].
In this situation, the surgical approach, which varies
depending on the pregnancy trimester is a particularly
concerning issue.
In this report, we discuss clinical considerations re-
lated to the unexpected finding of a large cystic lesion
that appears to be related to the pancreas in a pregnant
woman with acute abdomen.
Ethics
Written informed consent was obtained from the patient
for publication of this Case report and any accompany-
ing images. A copy of the written consent is available for
review by the Editor of this journal.
* Correspondence: jonarne.soreide@uib.no
1
Department of Gastrointestinal Surgery, Stavanger University Hospital,
N-4068 Stavanger, Norway
4
Department of Clinical Medicine, University of Bergen, Bergen, Norway
Full list of author information is available at the end of the article
© 2015 Søreide et al. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver
(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Søreide et al. BMC Surgery (2015) 15:119
DOI 10.1186/s12893-015-0106-1
Content courtesy of Springer Nature, terms of use apply. Rights reserved.

Case presentation
An otherwise healthy, non-obese 30-year-old woman,
para 0, at 12 weeks gestation was admitted to our hospi-
tal’s Department of Gastrointestinal Surgery with a
history of 3 weeks of varying but increasing pain in the
left upper quadrant of her abdomen and nausea without
vomiting. A palpable left subcostal mass was detected by
clinical examination, which revealed no other notable
findings except for her pregnant status. Ultrasound (US)
examination (Fig. 1) revealed a large cystic lesion (size,
17 × 10 cm) with septa related to the tail of the pancreas
and spleen. Magnetic resonance imaging (MRI) depicted a
multicystic lesion related to the pancreatic tail, between
the spleen and left kidney, with moderate dislocation of
both the kidney and spleen (Fig. 2). Routine biochemistry
was normal, and tumor markers carcinoembryomic anti-
gen (CEA = 4 μg/l), cancer antigen 125 (CA125 = 42kU/l),
carbohydrate antigen 19–9 (CA19–9 < 5 kU/l), and chro-
mogranin A(CgA= 2.8 nmol/l) were all within normal
ranges. The preoperative diagnosis was uncertain; how-
ever, based on imaging findings and the patient’syoung
age, a solid pseudopapillary epithelial neoplasm (SPEN)
was considered. A gynecologist evaluated the patient’s
pregnancy as normal. Relevant factors when considering
indications for surgery included the presence of increasing
clinical symptoms in a pregnant woman, the uncertain
nature of the rather large cystic pancreatic tumor, and the
possible risk and undesirable consequences of a rupture of
this tumor during the second or third trimester. In
accordance with the patient’s preferences, surgical treat-
ment was recommended. Open resection of the most dis-
tal portion of the pancreas, including the large cystic
tumor and spleen, was performed. Frozen sections of the
pancreas resection border confirmed that the resected
tissue was benign. The patient’s postoperative course was
uneventful, and she was able to leave the hospital on the
6
th
postoperative day without complications.
The multicystic tumor (Fig. 3) measured 19.5 × 10 × 7 cm
and exhibited close adherence to the distal pancreas but
without infiltration in the pancreas parenchyma or any
communication with the pancreatic ducts. Microscopically,
benign pancreatic tissue was confirmed, and the tumor was
multicystic with extremely thin septa, fibrosis and a
pattern of chronic inflammation, but no epithelial
tissue indicating that SPEN was unlikely. Additional
immunohistochemistry (IHC) with CD34 and CD 31
(Fig. 4) provided information to support a large-vessel
hemangioma lined with single endothelial layer without
cytologic atypia, and with focal degenerative changes in
septa and the cystic wall. This endothelial lining was
negative for cytokeratins and calretinin, excluding epi-
tehelial and mestothelial nature of the lesion Taken
together, the tumor morphological findings were char-
acteristic of a truly benign lesion most consistent with
adult pancreatic hemangioma, which was resected com-
pletely without any ruptures.
After surgery, the patient’s pregnancy proceeded un-
eventfully, and she spontaneously delivered a healthy child
Fig. 1 Ultrasound showed a large cystic lesion with septa and some sedimentation. Minimal Doppler signal of the large tumor (18 × 10 cm).
Spleen, pancreas and left kidney without focal lesions
Søreide et al. BMC Surgery (2015) 15:119 Page 2 of 6
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at the calculated gestational time. During follow-up of at
least 12 postoperative months, the patient has expressed
no complaints or concerns related to her treatment.
Discussion
While hemangiomas are rarely found in the pancreas,
they are very common in the liver. Mundinger et al.
reviewed the literature and found only 9 cases with con-
firmed adult pancreatic hemangiomas reported between
1939 and 2009 [21]. The lesions were most commonly
located in the head, or head/body of the gland, with a
size varying from 3 cm to 20 cm in diameter. Recently,
Bursics and coworkers [22] reported on another patient
(a 72-year-old man) surgically treated for a confirmed
adult hemangioma of the pancreas, and presented rele-
vant data on the 12 reported cases, including their own,
published until 2013. Male:female ratio was found to be
2:1, and the median age was 61 (range, 30–79) years. As
demonstrated by these 12 reported cases in the world
literature [21, 22], pain was the most common clinical
symptom which was also the case in our patient. The
pattern of a suggested cystic lesion by imaging was
Fig. 2 Coronal (a) and transversal (b) MRI T2 (FIESTA) view. Large tumor (T) medial to the spleen (S) and adjacent to the pancreatic tail dislocating the
left kidney (K) medial and cranial. Mostly high signal intensity at T2 and intermediate to high at T1, indicating cystic components with protein or blood. A
smaller part of the lesion had lower signal and diffusion restriction indicating more solid parts. (P) is the pregnant uterus. A solid pseudopapillaryepithel
neoplasia (SPEN) of the pancreas was suspected in this young woman with an encapsulated lesion with cystic, solid and hemorrhagic components
Fig. 3 Operative specimen with the cystic tumor and the spleen seen from the ventral (left) and the dorsal (right) side. The cystic tumor
measured 19.5 × 10 × 7 cm
Søreide et al. BMC Surgery (2015) 15:119 Page 3 of 6
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found in most patients with available information in
this regard. Notably, the CT patterns of a pancreatic
hemangioma are different from the CT signs of a liver
hemangioma, as a typical early peripheral contrast-
enhancement during the arterial phase is missing in the
former [23]. Therefore, this imaging modality is inef-
fective for ruling out pancreatic hemangioma.
Our patient was definitely in the younger range of age,
and importantly, we report on the first pregnant patient
diagnosed with an adult pancreas hemangioma. Although
the final morphologic diagnosis of this rare condition can
be challenging, CD 31 and CD 34 immunohistochemical
labeling adds valuable support to the diagnosis of a neo-
plasm of vascular origin [21].
Both benign and malignant pancreatic neoplasms are
rarely diagnosed during pregnancy; in particular, pancre-
atic cancer during pregnancy is extremely rare, with
fewer than 10 described cases to date [15, 16]. However,
a number of dilemmas can be encountered when
attempting to determine an accurate diagnosis to enable
appropriate treatment. Within this context, both US and
MRI are reliable and useful imaging modalities [5, 20]
without known health risks for the fetus [24]. When in-
dicated, CT can also contribute to imaging findings. Of
note, the risk that the fetus will develop congenital ab-
normalities due to the side effects of radiation exposure
(via repeat and/or multiphase CT scans) is considered to
be extremely low [25], nonetheless, due to concerns
regarding the potential for deleterious side effects, radi-
ation and various contrast agents should be limited and
used with care [24, 25].
Del Chiaro et al. [26] recently demonstrated that the
overall accuracy of preoperative diagnoses of cystic pan-
creatic lesions is only approximately 60 %, with similar
accuracies for asymptomatic and symptomatic lesions.
Thus, inaccurate preoperative assessments of pancreatic
cystic lesions are common; however, diagnostic errors
are clinically relevant in less than 10 % of these cases.
Data regarding lesion size, patient gender, and the
patient’s prior history (with respect to pancreatitis or
other relevant diseases) could be valuable information for
reaching appropriate decisions and thereby preventing the
overutilization of operative resection in patients with these
lesions [27]. For non-pregnant patients, additional im-
aging (e.g., contrast CT scans) and endoscopic ultrasound
(EUS)-guided aspiration of the cystic lesion for analyses of
DNA mutations and proteins within pancreatic cyst fluid
can contribute to the diagnostic workup [20, 28]. How-
ever, due to potential risks for a biopsy related bleeding,
rupture of the lesion or peritoneal seeding of biopsy
material, we did not regard our symptomatic pregnant pa-
tient with a large pancreatic lesion as a candidate for this
Fig. 4 aLarge dilated vascular structures lined by endothelial cells filled with red blood cells. The thickened vessel walls are composed of fibrous or
spindle cell stroma with mild chronic inflammation. (H and E; original magnification: ×10). bLarge dilated vascular structures lined by endothelial cells
filled with red blood cells. The thickened vessel walls are composed of fibrous or spindle cell stroma with mild chronic inflammation and hemosiderin
loaded macrophages (arrow) (H and E; original magnification: ×20). cMicroscopy showing large dilated vascular structure lined by endothelial cells.
The wall is infiltrated with mild chronic inflammation. (H and E; original magnification: ×100). d. Immunohistochemical expression of CD31 (brown)
depicting the endothelial lining of larger vascular spaces and small vessels in the stroma (original magnification, ×20)
Søreide et al. BMC Surgery (2015) 15:119 Page 4 of 6
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diagnostic approach. Her subjective and increasing com-
plaints, the uncertain nature of the lesion, and the risk of
tumor rupture with undesired side effects or complica-
tions later during her pregnancy, were all important con-
siderations for the timing of treatment. The preoperative
diagnosis of a possible SPEN was not definitively deter-
mined. As recently suggested [29] SPENs are rare cystic
lesions that frequently occur in young women and pa-
tients with these lesions exhibit good prognoses if radical
surgical resection can be achieved. Despite the challenging
nature of a definite diagnosis, the only feature of the de-
scribed case that could be associated with malignancy was
a large tumor size at diagnosis [20, 29]. Thus fare, malig-
nancy has not been reported for pancreatic hemangiomas.
Based on the rarity of this condition, the lack of reliable
follow-up data, and the sparse literature on this topic, an
understanding of the pathophysiology and natural history
of these lesions remains at an early stage [21].
Conclusion
Although reports have described a number of anecdotes
involving worrisome histories of pregnant women with
malignant tumors, including carcinomas of the pancreas
[16, 30, 31], pancreatic neuroendocrine tumors [14], and
bleeding neoplasms [32], or spontaneous SPEN rupture
[33], these cases are generally exceptions rather than the
rule. Nevertheless, given the objective of providing opti-
mal care to both mother and child, the determination of
an appropriate treatment schedule for any young pregnant
woman requires the consideration of a wide range of
diagnoses and a number of important factors. Although
the second trimester is considered to be the most favor-
able time for surgical intervention for pancreatic tumors
[15], multidisciplinary consultation should occur with
respect to a pregnant patient’s diagnosis, indications and
timing to ensure that the best possible outcomes for both
the mother and the child are achieved [15, 18, 19, 22, 26].
Abbreviations
CT: Computer tomography; EUS: Endoscopic ultrasound;
IHC: Immunohistochemistry; MCN: Mucinous cystic neoplasm; MRI: Magnetic
resonance imaging; SPEN: Solid pseudopapillary epithelial neoplasm.
Competing interests
The authors declare they have no competing interests.
Authors’contributions
Study conception and design: JAS. Data acquision: JAS, OJG, EG. Data
interpretation: JAS, OJG, EG. Drafting of the manuscript: JAS. Manuscript
editing and final approval: JAS, OJG, EG.
Acknowledgements
The authors appreciate the patient’s consent to present this case.
Author details
1
Department of Gastrointestinal Surgery, Stavanger University Hospital,
N-4068 Stavanger, Norway.
2
Department of Radiology, Stavanger University
Hospital, Stavanger, Norway.
3
Department of Pathology, Stavanger University
Hospital, Stavanger, Norway.
4
Department of Clinical Medicine, University of
Bergen, Bergen, Norway.
Received: 6 July 2015 Accepted: 19 October 2015
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