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Yu-Ping Gao, Jian-Shan Zhu, Wen-Jun
Zheng, Department of Pathology, Affiliated Renji Hospital, Shanghai
Second Medical University, Shanghai 200001, China
Correspondence to: Dr Yu-Ping Gao, Department of
Pathology, Affiliated Renji Hospital, Shanghai Second Medical University,
145 Shandong (c) Road, Shanghai 200001, China. jzmgyp@online.sh.cn
Telephone: +86-21-58752345 Ext. 3349
Fax: +86-21-58752345
Ext. 3349
Received: 2003-11-21 Accepted:
2004-01-15
Abstract
AIM: To analyze the
clinicopathological features of Brunner's gland adenoma of the duodenum.
METHODS: A rare case of Brunner's gland adenoma of the
duodenum was described and related literature was
reviewed.
RESULTS: Brunner's gland adenoma of the duodenum appeared
to be nodular hyperplasia of the normal Brunner's gland with an unusual
admixture of normal tissues, including ducts, adipose tissue and lymphoid
tissue. We suggested that it might be designated as a duodenal hamartoma
rather than a true neoplasm.
CONCLUSION: The most
common location of the lesion is the posterior wall of the duodenum near
the junction of its first and second portions. It can result in
gastrointestinal hemorrhage and duodenal obstruction. Endoscopic
polypectomy is a worthy treatment for benign Brunner's gland adenomas, as
malignant changes in these tumors have never been proven.
Gao YP, Zhu JS, Zheng WJ. Brunner's gland adenoma of duodenum:
A case report and literature review. World J Gastroenterol 2004; 10(17): 2616-2617
http://www.wjgnet.com/1007-9327/10/2616.asp
INTRODUCTION
Brunner's gland
adenoma, also known as Brunneroma or polypoid hamartoma, is a rare,
benign, proliferative lesion arising from the Brunner's glands of the
duodenum, accounting for 10.6% of benign tumors of the duodenum. The time
patients are usually asymptomatic and lesions are discovered incidentally.
These lesions manifest occasionally as a rare cause of duodenal
obstruction or upper gastrointestinal hemorrhage, and require surgical
excision[1]. This article reports a case of Brunner's gland
adenoma, and reviews briefly its clinical presentations, pathological
features and therapy.
CASE REPORT
A 32-year-old Chinese man complained of two episodes of
melenas and a two-month history of vague epigastric discomfort. The
patient had in October 2001 his first tarry stool episode without nausea,
vomiting and epigastralgia. Endoscopic examination revealed a mild ulcer
in the duodenal bulb. The symptoms were relieved by antacids and H2
blockers. He reported however another tarry stool in September 2002 and
complained about epigastric pain without nausea and vomiting during the
preceding 2 mo. Antacids were taken and the pain was relieved. Vital signs
were normal and no anemia was reported when he was admitted. The physical
examination had no unremarkable finding. The abdomen was soft without
palpating pain and jumping pain.
However, X-rays barium radiological examination of the upper
gastrointestinal in October 2002 revealed a nodular, polypoid-filling
defect mass measuring 3 cm�2.5 cm with a smooth surface and no ulceration
in the duodenal bulb. A tumor
was suspected. Follow-up endoscopy disclosed a lobulated, red-color tumor,
occupying the anterior wall of the bulb. The surface of the tumor was
smooth with mild depression at the top. The mucosa had no erosions and
ulcers. Multiple biopsy specimens were taken and interpreted as "mucosal
mild-medium atypia." CT scan was negative. A preoperative diagnosis of
malignant tumor was made and the patient prepared for operation. On
October 25, 2002, at the time of operation, a pedunculated polyp on a
short broad-based stalk, 3.5 cm�3 cm�2 cm in size, was found in the
anterior wall of the duodenal bulb. The common bile duct was normal. The
lesion was amputated at the base of the stalk.
The resected specimen showed a lobulated, polypoid mass
measuring 3.3 cm�2.5 cm�2.5 cm projecting into the duodenum. The stalk was
measured 1.8 cm in diameter and 0.5 cm in length. The tumor was completely
enveloped by the intact thin duodenal mucosa. The surface of the tumor was
smooth without erosions and ulcers. The cut surface of tumor had a
gray-red or gray-yellow color, revealing lobules. The consistency was
moderate. On microscopic examination, the tumor was composed of
hyperplasia of Brunner's glands. The hyperplasia formed lobules that were
separated by intervening bands of fibrous tissue, adipose tissue, ducts
and well developed aggregated lymphoid. No sign of malignancy was found in
the hyperplasia, Brunner's glands as well as the surrounded duodenal
mucosa. The frozen sections and the final pathologic diagnosis were
assessed as Brunner's gland adenoma. The patient had an uneventful
postoperative course and was discharged on the tenth postoperative day. He
has remained symptom free ever since and no episode of recurrent melenas
has been reported.
DISCUSSION
Besides the duodenal gland,
the duodenum has Brunner's glands under the mucin. Its structure and
function are similar to glands of the pylorus. Brunner's glands secrete an
alkaline fluid composed of viscous mucin, whose function appears to
protect the duodenal epithelium from acid chyme of the stomach. Brunner's
glands consist of submucosal mucin-secreting glands located exclusively in
the duodenum. They extend from the pylorus distally for a variable
distance, usually stopping at the first and second portions of the
duodenum, and less often stopping at the third and fourth portions.
In 1688 Brunner gave a precise anatomic description of the
duodenal submucosal glands and coined the term "pancreas secundarium." In
1846 Middeldorpf correctly identified these glands as a separate entity,
which he proposed be named Brunner's glands. Salvioli reported the first
adenoma of Brunner's gland in 1876. Since then, 150 cases or so have been
reported in literature of English language[2].
The etiology of Brunner's gland adenoma remains obscure. It
tends to present predominantly in the fifth and sixth decades of man's
life with no sex predominance. It has been found although the size of
adenoma might extend from 1-12 cm, it is generally 1-2 cm in
diameter[3]. The most common location for the lesion is the
posterior wall of the duodenum near the junction of its first and second
portions. Brunner's gland adenoma was rarely found extending to the
proximal jejunum[4].
Brunner's gland adenoma has fallen into two categories:
symptomatic tumors and asymptomatic ones that are only found incidentally.
Symptomatic tumors can further be divided into hemorrhagic and obstructive
tumors. The clinical manifestations of the former are gastrointestinal
hemorrhage, due to ulceration or erosion of the tumor. Obstructive tumors
occur when hyperplasia diffuses or a single adenoma grows too large,
causing epigastric bloating, discomfort, vomiting or weight loss. Duodenal
intussusception has been reported only in two patients[2],
probably because of the fixation of duodenum to the posterior abdominal
wall. There are also reports about patients who complained of diarrhea
owing to duodenal motor disturbances[5].
Preoperative histological diagnosis at present is not always
easy. In X-rays barium examination, the findings are often nonspecific
because there is usually a sessile or pedunculated polypoid-filling defect
in the duodenal bulb. Some doctors hold that hypotonic duodenography
should play a vital role in establishing the diagnosis and should be
treated as the best method to check the surface of the lesion. Endoscopy
has an additional function in diagnosing and treating Brunner's gland
adenoma, since it can verify the histological diagnosis and remove the
tumor simultaneously. Endoscopic pinching biopsy however usually gave a
negative result because the tumor was almost covered entirely with thick
intact duodenal mucosa in the biopsy sites and the biopsy was often not
deep enough to reach the submucosal tumor tissue[6]. In our
case, the endoscopic biopsy was negative, and the condition was diagnosed
as chronic gastritis, although the final pathologic diagnosis indicated
that the patient suffered from Brunner's gland adenoma. CT examination
appeared to be unrevealing.
Pathomorphological features of
Brunner's gland are characterized by the presence of nondysplastic,
lobulated Brunner's glands. Its hyperplasia is divided into diffuse
hyperplasia, nodular hyperplasia and adenomatous hyperplasia with or
without erosion or ulcer. In our opinion, the unusually admixture of
normal tissues, including Brunner's glands, ducts, adipose tissue, and
lymphoid tissue, supports the designation of these lesions as a hamartoma
or nodular hyperplasia rather than a true neoplasm. It is a tumor without
malignant predisposition. The malignant type is rare. Fujimaki et al.
reported recently one patient with a focus atypical
gland[7].
It is still controversial whether asymptomatic Brunner's gland
adenoma found incidentally needs surgical removal. Some people think that
it needs no treatment, whereas others hold that it should undergo
endoscopic excision in order to prevent complications. There have been
several reports[2,4] that Brunner's gland adenoma could give
rise to acute profuse bleeding, which results in shock of patients.
Symptomatic Brunner's gland adenoma, in our point of view, usually needs
surgical treatment. When the tumor is small or pedunculated, endoscopic
polypectomy is the first choice. Open surgical excision is reserved for
cases where snaring has failed or when tumor is too large. The outcome of
operation is usually excellent and there is no recurrent ever
reported.
REFERENCES
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T, Fujishima M. A large Brunner's gland adenoma removed by
endoscopic
polypectomy. Endoscopy
1990; 22: 192-193
2 Peetz ME, Moseley HS.
Brunner's gland hyperplasia. Am Surg 1989; 55: 474-477
3 Nakanishi T, Takeuchi T, Hara
K, Sugimoto A. A great Brunner's gland adenoma of the duodenal bulb. Dig
Dis Sci
1984; 29: 81-85
4 Levine JA, Burgart LJ, Batts
KP, Wang KK. Brunner's gland hamartomas: clinical presentation and
pathological features
of 27 cases.
Am J Gastroenterol 1995; 90: 290-294
5 Spellberg MA, Vucelic B. A
case of Brunner's glands hyperplasia with diarrhea responsive to
cimetidine. Am J
Gastroenterol
1980; 73: 519-522
6 Gourtsoyiannis NC,
Zarifi M, Gallis P, Mouchtouris A, Livaditou A. Radiologic appearances of
Brunner's gland adenoma:
a case
report. Eur J Radiol 1990; 11: 188-190
7 Fujimaki E, Nakamura S, Sugai T,
Takeda Y. Brunner's gland adenoma with a focus of p53-positive atypical
glands. J
Gastroenterol 2000; 35:
155-158
Edited by Qiu WS and Wang XL Proofread by Xu FM
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