Department of Pathology, Jackson Memorial Hospital/University of Miami, Leonard M. Miller School of Medicine, Miami, FL, USAUnited States
Department of Pathology, Cleveland Clinic Florida, Weston, FL, USAUnited States
Division of Gastroenterology, Jackson Memorial Hospital/University of Miami, Leonard M. Miller School of Medicine, Miami, FL, USAUnited States
Division of Gastroenterology, Cleveland Clinic Florida, Weston, FL, USAUnited States
Also available as PDF (figures and tables appear in the PDF).
Objective: Brunner gland cysts are rare with only 14 cases reported in the literature. The term has been used to consolidate the following entities: Brunner’s gland cyst, cystic Brunner’s gland hamartoma, Brunner’s gland cystadenoma and mucocele of the Brunner gland. Methods: We present the clinico-pathological features of two cases of Brunner gland cyst along with a review of the literature. Results: Brunner gland cyst affects slightly more men than women. The age range is 30 to 72 years with an average of 55 years. The lesions range from 1 to 5 cm in size, the average being 2.3 cm in greatest dimension. The majority of lesions are found incidentally in patients with symptoms not specifically related to the lesion. They affect predominantly the first and second portion of duodenum. The majority of the lesions are single and pedunculated filling the sub mucosa. They can be unilocular or contain multiple cysts divided by fineseptae. The epithelial lining is of columnar and clear cells with basally located nuclei. Conclusion: Brunner gland cysts are benign lesions that are usually not diagnosed pre-operatively. The cases described here add awareness of this entity among pathologists and gastro enterologists allowing for a better recognition of this rare entity.
Keywords: Duodenum; Brunner gland; Cystadenoma; Cyst; Hamartoma
Cystic lesions of the Brunner gland are rare. They have been referred to as Brunner’s
gland cyst, cystic Brunner’s gland hamartoma, Brunner’s gland cystadenoma and mucocele of
the Brunner gland. Recently these entities have been grouped under the term Brunner gland
cyst. Even with these combined entities, there are only fourteen Brunner gland cysts reported
in the English literature. We present two new cases of Brunner gland cyst and compared them
to those previously reported. Their clinical presentation and similarities are summarized.
A 69-year-old Hispanic male was referred by his primary care physician to our institu-
tion for further studies of a duodenal lesion that was discovered after a workup for acid reflux.
Endoscopic ultrasound showed a 17x6 mm septated cyst that appeared to arise from the sub-
mucosa of the second portion of the duodenum (Figures 1 and 2). No other abnormalities were
The specimen consisted of an ovoid lesion with a 0.5 cm cystic cavity. Microscopic
examination revealed a submucosal multicystic lesion lined by tall, columnar cells with round
to those seen in the adjacent normal Brunner glands. In other ar-
eas, the lining cells were smaller with pink cytoplasm. The cysts
were filled with a serous-like fluid. Necrosis, mitotic activity,
and nuclear atypia were absent. The cells contained neutral, Pe-
riodic Acid Schiff (PAS) positive mucin, similar to the adjacent
Brunner’s glands
The second patient is a 52-year-old Caucasian man re-
ferred to our institution because of epigastric pain and a duode-
nal mass found at endoscopy. Another endoscopy was performed
along with endosonography. An intramural (subepithelial) lesion
was found in the second portion of the duodenum. The lesion
appeared to originate from within the submucosa (layer 3). The
possibilities of pancreatic rest or Brunner’s gland hyperplasia
were considered. The lesion was hypoechoic, heterogeneous and
multicystic that measured 14.5 mm x 10.9 mm. The outer mar-
gins were well defined. An intact interface was seen between the
mass and the adjacent structures suggesting a lack of invasion.
An endomucosal resection of the mass was performed.
Histologically, the multicystic lesion was located in the
submucosa of the duodenum and was composed of tall cells with
basally located round nuclei. The cytoplasm was clear and abun-
dant. In areas, there were aggregates of glands by the single row
of epithelial cells lining the cysts creating a nodular configura-
tion. No mitosis, necrosis or atypia was observed.
denum in 1933.1 Varnholt, et al. were the first to group Brunner
gland cyst, Brunner cyst, mucocele of Brunner gland, and cystic
Brunner’s gland hamartoma as one entity.2 Later, Powers, et al.
added Brunner gland cystadenoma to this group.3 There are four-
1-12 teen cases reported in the English literature.1-12 Information from
these cases and the current two is summarized in Table 1.
Brunner gland cysts are benign, as evidenced by the
fact that the lesion described by Golan, et al. was present for
15 years without any long-term consequences.9 They are widely
believed to be retention cysts that develop after obstruction of a
larger duct of the Brunner gland outflow tract.2
Based on this case and the well-described fourteen pre-
viously reported cases, the following conclusions can be made.
The lesion can occur throughout the duodenum, specifically any
place that contains Brunner glands. The large majority are single
lesions with one patient reported to have two.3 Most of the le-
sions are pedunculated and located in the submucosa, but ses-
sile lesions have been reported and one was transmural.8 While
some are unilocular, others contain multiple cysts divided by
fine septae. The cysts are lined by mucinous, columnar cells with
basally located nuclei, resembling normal Brunner gland cells.
Eosinophilic cells lining the cyst wall are also common. Only
the case described by Chatelain, et al. showed ciliated columnar
7 10
Moul, et al. (cur- Brunner 2015 69, M ing in a patient portion of 1.7 x 0.7 x 0.3 Sessile Sub-mucosa Single rent case) gland cyst
Moul, et al. Brunner 2015 52, M Epigastric pain portion of (current case) gland cyst
4 Brunner 2011 Galiatsatos 72, M Incidental second gland cyst
Cystic Brun- Third por- 5 tory of nausea, 2009 Park, et al. ner’s gland 30, M tion of the
hamartoma duodenum
3 Brunner Dyspepsia and 2008 Powers, et al. 46, F the second gland cyst odynophagia
3 Brunner 2008 Powers, et al. 67, F unexplained part of the gland cyst
3 Brunner 2008 Powers, et al. 59, F abdominal bulb (first gland cyst
2 Brunner being treated Not speci- 2007 Varnholt, et al. 41, F 1.1 x 0.9 x 0.6 Sessile Sub-mucosa Single gland cyst for H. pylori- fied
Cystic Brun- 6 duodenum 2003 Yamakawa, et al. ner’s gland 64, F Epigastric pain 2.4 x 1.1 x .5 Pedunculated Sub-mucosa Multiple epithelium ;
hamartoma
Brunner
gland
hamartoma
with pre- Duodenal 7 Two-day history 2002 Chatelain, et al. dominant 43, M bulb (first
adipose portion)
tissue and
ciliated
cysts
Mucocele 8 quadrant pain Not speci- 1980 Fisher of Brunner 45, F Not specified Not specified Sub-mucosa Single dilated glandular
gland
Cystic Brun- First part of 9 bleeding; had a 1978 Golan J, et al. ner’s gland 64, M the duode-
hamartoma num.
Incidental find- Second Focal eosinophilic
cells with acid reflux duodenum
Second Focal eosinophilic 1.4 X 1.0 Polypoid Sub-mucosa Single cells duodenum
Normal appearing
duodenal mucosa Distal end and dilated lymof the phatic channels 2 Polypoid Not specified Not specified and capillaries part of the (pictures not duodenum available for
review)
Lobular collec- Three-day histion of mature
4 x 3 Pedunculated Sub-mucosa Multiple Brunner’s glands, vomiting, and multifocal cystic epigastric pain.
dilation.
Two lesions Cystic spaces
located in lined by hy-
1.8 and 2.2 Sessile Sub-mucosa Single perplastic cells
part of the similar to Brunner
duodenum glands cells.
Incidental in Cystic spaces
a patient with Second lined by hy-
1.5 Sessile Sub-mucosa Single perplastic cells
iron-deficiency duodenum similar to Brunner
anemia glands cells.
Incidental in Cystic spaces
a patient with Duodenal lined by hy-
1.0 Nodule Sub-mucosa Single perplastic cells
pain, heartburn, portion) similar to Brunner
steatorrhea glands cells.
Incidental find-
ing in a women Cyst lined by a
simple cuboidal-
to-columnar
associated epithelium.
gastritis.
Multilocular cysts Descending lined by columnar
(second dilated ductal portion) structures.
Prominent mature
adipose tissue, Multiple, 3.5 Pedunculated Sub-mucosa hyperplastic lobof regurgitation. ciliated ules of Brunner
glands
Intact duodenal
mucosa with Right upper prominently
intermittently fied spaces, one for two months being large and
cystic.
Cysts were lined Acute gasby columnar and trointestinal cuboidal epithe-
5 x 4 x 2 Pedunculated Sub-mucosa Multiple lium. The surface known duodeepithelium was nal polyp for 15 partly pyloric and years partly duodenal.
10 Brunner’s vomiting, and Taura M, et al. 54, F bulb (first cyst epigastric pain
Brunner’s 11 20 lb weight duodenum, Wolk DP, et al. gland cysta- 68, M
denoma
12 Brunner’s Hately 54, M tent vomiting, the duodegland cyst
Simple,
Rankin and multilocular 1 54, M anemia and - Newell cystade-
noma
Table 1. Brunner gland cysts reported in the English literature: Patient characteristics, clinical presentation, diagnostic modality and lesion location, and histological characteristics.
Ultrastructural studies performed by Taura, et al. in 1977 dem-
onstrated epithelial cells containing membrane-bound secretory
granules in the cytoplasm, mainly in the apical region, and a
well-developed Golgi apparatus. The luminal surfaces contained
microvilli. These features suggested that the cells of Brunner
gland cysts were functionally more active than normal Brunner
gland cells. Another distinct histological feature was the lack of
neuroendocrine cells within the Brunner gland cysts, which is
not true of normal Brunner’s glands.10
There is no sex predilection; fifty-three percent were
male. The age range was 30 to 72 years of age, with the aver-
age being 55 years. The lesions ranged from 1 to 5 cm in size,
the average being 2.3 cm in greatest dimension. While increased
size correlated with more clinical symptoms, the patient with the
largest reported lesion was not symptomatic from the lesion but
from an adjacent ulcer.9 Nonetheless, most clinical symptoms
are regurgitation, vomiting, and epigastic pain. However, it may
be discovered incidentally for unrelated symptoms. Most often,
the lesions are located in either the first or second portion of the
duodenum. This corresponds to the most common locations of
the Brunner’s gland, which are mostly concentrated in the first
portion and gradually decrease in number throughout the length
of the duodenum.13
The differential diagnosis on endoscopic imaging in-
cludes duplication cysts, lipomas, neuroendocrine tumors and
Brunner gland hamartomas. Using endoscopic ultrasound it is
possible to differentiate the echostructure (cystic, solid, hypo-
or hyperechoic) and wall layer of involvement. The differential
diagnosis of hypoechoic/anechoic duodenal lesions would be
mainly duplication cysts, stromal cell tumors and neuroendo-
crine tumors. Our first patient underwent resection to ensure his
was not a cystic neuroendocrine lesion. Histologically the main
differential diagnosis is Brunner’s gland hamartoma, formerly
Lined by tall
columnar cells
Nausea, with basal nuclei.
Duodenal Multinucleated 1.5 Not specified Sub-mucosa Single cells were interportion) for several days mingled with the
epithelial lining
cells.
Third por- Multiple fluid-filled
Presented with tion of the cystic spaces
confined to the 3 x 3 Not specified Sub-mucosa Multiple loss and eruc- located on submucosa,
tation. the poste- lined by Brunner
rior wall glands
Cyst lined by co- Two year hislumnar epithelium tory of intermit- First part of and had a direct 1.5 Sessile Sub-mucosa Single origin from one now after every num.
of the Brunner meal.
glands.
18 m history
of pernicious Cyst lined by
2 Not specified Sub-mucosa Multiple cuboidal epithelial
ulcer-like cells.
dyspepsia.
known as Brunner’s gland adenoma. These lesions are admix-
tures of Brunner’s glands, ducts, adipose tissue and lymphoid
tissue. While their ducts may be dilated, cystic lesions are not
characteristic of this entity.14
Brunner gland cysts are rare benign lesions of the duo-
denum. The treatment is surgical excision or polypectomy. In-
creased awareness of these lesions helps for a better recognition
of this entity.
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above.