Case Report
Open Access
A Pop Eye Mucosal Bulge in the Duodenal Bulb
Tian-Yin Chen1, Xiao-Cen Zhang1, Ming-Yan Cai1, Xiao-Wen Ge2, Ping-Hong Zhou1*
1Endoscopy Center and Endoscopy Research Institute, Zhongshan Hospital, Fudan University, Shanghai,
China
2Pathology Department, Zhongshan Hospital, Fudan University, Shanghai, China
2Pathology Department, Zhongshan Hospital, Fudan University, Shanghai, China
*Corresponding author: Ping-Hong Zhou, Endoscopy Center and Endoscopy Research Institute, Zhongshan Hospital, Fudan University, 180
FengLin Road, Shanghai, 200032, P. R. China. Phone: (+86)-21-64041990. E-mail:
@
Received: March 09, 2016; Accepted: March 23, 2016; Published: May 03, 2016
Citation: Chen TY, Zhang XC, Cai MY, Ge XW, Zhou PH (2016) A Pop Eye Mucosal Bulge in the Duodenal Bulb. Gastroenterol Pancreatol
Liver Disord 3(3): 1-2. http://dx.doi.org/10.15226/2374-815X/3/3/00158
Abstract
Neoplasms and malignancies are seldom seen in the duodenum
in comparison with other parts of the alimentary tract. Duodenal
subepithelial lesions with umbilication are rare, often regarded
typical for ectopic pancreas. Meanwhile, premalignant/ malignant
transformation of ectopic gland might even be rarer. We present a case
of a duodenal intraepithelial neoplasia, most likely originated from
an ectopic pancreas, hidden under normal epithelium of duodenum;
with a "pop eye" left on it's the surface.
Keywords: Duodenal ectopic pancreas; Intraepithelial neoplasia
Keywords: Duodenal ectopic pancreas; Intraepithelial neoplasia
Introduction
Neoplasms and malignancies in the duodenum are relatively
rare in comparison with other parts of the alimentary tract.
Duodenal subepithelial lesions usually include ectopic pancreas,
lipomas, gastrointestinal stromal tumors, lymphoma and etc.
Non-ampullary lesions present with inconspicuous symptoms
and are often discovered during a routine endoscopy, while
ampullary lesions may have an early onset of jaundice and
pancreatitis due to the obstruction of the duodenal papillary
orifice. Endoscopic techniques such as high-definition white-light
endoscopy (HWE), narrow band imaging (NBI), and confocal laser
endomicroscopy (CLE) have been applied to detect alimentary
lesions and distinguish from cancerous tumors [1]. We report a
case of a duodenal tubulovillous adenoma, probably originated
from an ectopic pancreas, presenting with an appearance of pop
eye mucosal bulge.
Case Report
Upper GI endoscopy in a 58-year-old man who complained
of chronic upper abdominal discomfort revealed a 2.0cm bulge
in the duodenal bulb (Figure 1). There were no specific findings
in his past and family history. Laboratory analysis came back
negative. Abdominal CT scan ruled out lesions or metastasis
in other places. The bulged lesion had smooth surface mucosa
and an orifice oozing clear fluid at the top. It was later removed
with endoscopic piecemeal mucosal resection (EPMR), and the
specimen had a jelly-like texture (Figure 2). HE section showed
tubulovillous adenoma with middle-high grade dysplasia,
oxyphilic type (Figure 3). Immunohistology showed KI67 (30%+), MUC5 (+), MU6 (+), CK7 (+), CK20 (-) (Figure 3), leading
to a diagnosis of intraepithelial neoplasia originating from ectopic
gland (most likely biliary-pancreatic duct). The patient had no
fever, abdominal infection, pancreatitis or other complications
after EPMR and was discharged after 3 days.
Figure 1:The lesion presented with a 2.0cm bulge in the duodenal bulb.
The bulged lesion had smooth surface mucosa and an orifice oozing
clear fluid at the top.
Figure 2:The lesion was resected by endoscopic piecemeal resection
and the specimen had a jelly-like texture.
Figure 3:(a) HE section showed tubulovillous adenoma with middlehigh
grade dysplasia, oxyphilic type. (b) Immunohistology showed KI67
(30 %+), (c) MUC5 (+), (d) CK20 (-).
Discussion
Duodenal subepithelial lesions are rare, with majority
probably being neuroendocrine tumors [2]. Subepithelial
lesions with umbilication are also rare, often regarded typical
for ectopic pancreas [3]. Meanwhile, premalignant/ malignant
transformation of ectopic gland might even be rarer [4]. Our case
combined all three scenarios. The pop eye mucosal bulge with an
umbilicus seemed to be a subepithelial lesion, whose diagnosis
was most likely an ectopic pancreas according to its appearance
before resection. However, HE staining of the specimen showed
tubulovillous adenoma with middle-high grade dysplasia and
finial pathological diagnosis was intraepithelial neoplasia
originating from ectopic gland.
The reported incidence of ectopic pancreas ranges from 0.25% to 1.2% [5], mostly found in duodenum (29.3%), stomach (27.4%), jejunum (15.7%), ileum (5.9%), Meckel's diverticulum (5.1%), and gallbladder (2.7%) [6]. Chances are that similar pathological changes such as acute pancreatitis or cyst formation may occur in ectopic pancreas, but dysplasia and malignant degeneration rarely develops [7]. In our case, the intraepithelial neoplasia was hidden under normal epithelium of duodenum; with a "pop eye" left on it's the surface. Without the resection, it might never be suspected and have a chance of malignancy, causing much more serious problems to the patient.
Accurate diagnosis of subepithelial tumors before resection remains an arduous problem. Endoscopic ultrasound (EUS) does not contribute to the differential diagnosis as well in subepithelial tumors as in epithelial lesions. Tissue biopsy and sampling before operation are also ineffective due to their high false negative rate [8]. Thus, best treatment for duodenal subepithelial lesions is unclear due to the limited experience. However, our case highlights the importance of endoscopic intervention, rather than pure observation and surveillance, given the malignant potential of a seemingly benign lesion.
The reported incidence of ectopic pancreas ranges from 0.25% to 1.2% [5], mostly found in duodenum (29.3%), stomach (27.4%), jejunum (15.7%), ileum (5.9%), Meckel's diverticulum (5.1%), and gallbladder (2.7%) [6]. Chances are that similar pathological changes such as acute pancreatitis or cyst formation may occur in ectopic pancreas, but dysplasia and malignant degeneration rarely develops [7]. In our case, the intraepithelial neoplasia was hidden under normal epithelium of duodenum; with a "pop eye" left on it's the surface. Without the resection, it might never be suspected and have a chance of malignancy, causing much more serious problems to the patient.
Accurate diagnosis of subepithelial tumors before resection remains an arduous problem. Endoscopic ultrasound (EUS) does not contribute to the differential diagnosis as well in subepithelial tumors as in epithelial lesions. Tissue biopsy and sampling before operation are also ineffective due to their high false negative rate [8]. Thus, best treatment for duodenal subepithelial lesions is unclear due to the limited experience. However, our case highlights the importance of endoscopic intervention, rather than pure observation and surveillance, given the malignant potential of a seemingly benign lesion.
Authors' Contributions
TY Chen and XC Zhang contributed the same in the writing
of the manuscript. MY Cai was involved with formation of
the study concept and design, drafting of the manuscript and
the literature review. XW Ge contributed to the pathological
and immunohistochemistry examinations. PH Zhou carried
out the endoscopic operation on the patient and was the main
contributor in the writing of the manuscript. All authors have
read and approved the final version of the manuscript.
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