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Giant Brunneroma
 
Anita B Shah, Girish A Muzumdar, Arun R Chitale
 
A giant Brunneroma occurring as a polypoidal mass in the second part of duodenum is reported. The patient was a 35 year old woman who presented with complaints of haematemesis and melaena. The polyp measured 5.0 x 3.0 x 2.5 cm. Microscopically it was composed of conglomerates of closely set Brunner’s glands.
 
Introduction

Brunner’s glands are normally situated in the duodenal submucosa and occasionally extend above the muscularis mucosae into the lamina propira. Hyperplasia of Brunner’s glands exists in three forms a) diffuse glandular proliferation imparting a coarse nodularity to the duodenal mucosa, b) limited discrete nodules in the proximal duodenum and c) solitary nodules which are termed adenomas (Brunneroma). These are generally encountered as incidental findings during upper gastro-intestinal endoscopy.

The vast majority of Brunneromas are small, ranging in size from 2.0-3.5 cm. However, to our knowledge, this is the largest such adenoma described.

 
Case Report

Clinical features : A 35 year old housewife presented with complaints of haematemesis off and on since a year and three episodes of melaena since four months. Upper gastro-intestinal endoscopy revealed a large pedunculated polyp in the second part of the duodenum. CT-scan showed no evidence of extra-duodenal extension of the lesion. The polyp was surgically excised.

Pathological findings : The firm nodular polyp measured 5.0 x 3.0 x 2.5 cm. The outer surface was smooth and glistening gray-white (Fig. 1). Cut surface showed fleshy homogeneous tan tissue. Microscopically, the polyp was entirely composed of closely set conglomerates of normal appearing Brunner’s glands. No nuclear atypia was detected. The overlying villous duodenal mucosa was intact.

Fig. 1 Fig.2
 
Discussion

Hyperplasia of Brunner’s glands presenting as solitary nodules are termed “adenomas” or “Brunneromas”. The vast majority are incidentally detected as tiny nodules during upper gastro-intestinal endoscopy.1

The first case was described by Cruveilhier in 1835 and termed a “hamartoma”. This patient had a fatal duodenal intussusception. Subsequently, 143 cases have been described in literature of which only 25 nodules were > 2.0 cm in diameter.2

In a study of 27 cases, Levine et al showed that there was no sex predilection and the patients presented in the fifth to sixth decades. All “tumours” were removed either surgically or endoscopically and the outcome was uniformly favourable. Even after a seven year follow-up there was no additional morbidity.3

Most patients present with abdominal pain, vomiting, haemorrhage and obstruction.4-6 The symptomatology depends upon the location of the lesion i.e. haemorrhage occurred in patients with nodules in the second part of the duodenum, whereas abdominal pain was more frequent if the lesion was in the first part.7 In our case the polyp presented with haematemesis and melaena, being situated in the second part of the duodenum.

The distinction between hyperplasia and “adenoma” is arbitrary and based entirely on the endoscopic appearance. No case of Brunner’s gland carcinoma has been reported and there is no substantial evidence that these are neoplastic entities. Hence, the term Brunner’s gland nodule is now preferred.1

Anecdotal cases of “Brunneromas” displaying histological variations have been described. Chatelain et al described a 3.5 cm nodule in the first part of the duodenum containing predominantly adipose tissue and cysts lined by ciliated epithelium.8 Fujimaki et al reported a 2.2 cm “tumour” in the duodenal bulb of a 43 year old man. It had a focus of “atypical” cells within the Brunner gland adenoma. Immunohistochemically, MIB-1 and p53 positivity were demonstrated, representing a probable neoplastic lesion. However, true neoplasms in these lesions are indeed rare and only one other case associated with micro-carcinoid tumour has been reported.9

 
 
References
1. Sternberg SS. “Diagnostic surgical pathology” third edition. Lippincott Williams and Wilkins Pages 1360-1.
2. Chen TM, Changchien CS, Hsu CC, Hu TH. Brunner’s gland hamartomas : Report of three cases. Changgeng Yi Xue Za Zhi 1999; 22 (2) : 271-6.
3. 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 (2) : 290-4.
4. Varma D, Prakash K, Augustine P, et al. Brunner’s gland adenoma with circumferential duodenal involvement. Indian J Gastroenterol 2001; 20 (6) : 243-4.
5. Yadav D, Hertan H, Pitchumoni CS. A giant Brunner’s gland adenoma presenting as gastro-intestinal hemorrhage. J Clin Gastroenterol 2001; 32 (5) : 448-50.
6. Cavallaro G, Albanese V, Taranto F, et al. Brunner’s adenoma, esophageal reflux and gastric ulcer. A case report. Chir Ital 2000; 52 (6) : 703-6.
7. Adeonigbagbe O, Lee C, Karowe M, et al. A Brunner’s gland adenoma as a cause of anemia. J Clin Gastroenterol 1999; 29 (2) : 193-6.
8. Chatelain D, Maillet E, Boyer L, et al. Brunner gland hamartoma with predominant adipose tissue and ciliated cysts. Arch Pathol Lab Med 2002; 126 (6) : 734-5.
9. Fujimaki E, Nakamura S, Sugai T, Takeda Y. Brunner’s gland adenoma with a focus of p53 positive atypical glands. J Gastroenterol 2000; 35 (2) : 155-8.