An Unusual Cause of Duodenojejunal Intussusception and Melena

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• Answer to the Clinical Challenges and Images in GI Question: Image 4: Brunner's Gland Hamartoma With Low-Grade Dysplasia
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Question: A previously healthy 58-year-old woman was referred to our hospital with symptoms of an intestinal obstruction, related to duodenojejunal intussusception. Three weeks before admission, she had experienced an episode of melena, and hematologic investigations revealed anaemica, with a hemoglobin concentration of 6.8 g/L. Upper gastrointestinal endoscopy revealed an ulcerated duodenal mass, for which mucosal biopsies were nondiagnostic.

Computed tomography and magnetic resonance imaging (MRI) were performed. Figure A (axial contrast-enhanced T1-weighted gradient-echo MRI) shows duodenojejunal intussusception (arrowhead) with a heterogeneously enhancing mass serving as a lead point (Figure B, arrows).

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The patient underwent laparotomy with a presumed diagnosis of gastrointestinal stromal duodenal tumor. Through a longitudinal duodenotomy, a long, reddish, pedunculated, ulcerated polyp was found in the anterior wall of the duodenal bulb, protruding into the proximal jejunum. The polyp was removed with wedge duodenal resection, and the postoperative recovery was uneventful.

The polyp was covered with a smooth mucosa with an ulceration at the apex. Figure C shows a cross-cut of the specimen, with a 5-cm well-circumscribed, lobulated mass, with a pink-tan appearance, and multiple cystic spaces (up to 2 cm). Figures D and E show hematoxylin and eosin staining of the microscopic section.

What is the diagnosis?

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Answer to the Clinical Challenges and Images in GI Question: Image 4: Brunner's Gland Hamartoma With Low-Grade Dysplasia 

Microscopic examination showed that this mass was covered with an ulcerated duodenal mucosa, and composed of lobules of relatively bland appearing Brunner's glands, which were surrounded by bundles of fibromuscular and connective tissue. Some ducts were dilated and lined by a papillary structures (Figure D). Moreover, throughout the specimen there were foci of obvious low-grade dysplasia (Figure E). There were no high-grade dysplasia or invasive carcinoma.

Brunner's glands are mucosal and submucosal alkaline-secreting glands that are most commonly located in the duodenum. Their mucinous secretions buffer the acidic chyme entering from the stomach. Brunner's gland hamartomas (BGH) are rare, benign small bowel tumors of unknown pathogenesis. They account for approximately 5% of all duodenal masses and occur most commonly in the 5th and 6th decades of life, with no gender or race predominance. The most common location is the duodenal bulb (70%), followed by the second portion of the duodenum (26%), and the third one (4%) representing normal distribution of these glands.¹, ² They usually range from 1 to 6 cm in diameter. Although commonly an incidental finding, clinical presentation of BGH is variable; obstruction and gastrointestinal bleeding are the most common clinical manifestations. Rare cases of obstructive jaundice, recurrent pancreatitis, and intussusception, as the case herein presented, have been reported. Endoscopy reveals a submucosal mass; therefore, standard mucosal biopsies are of little benefit.¹, ² On endoscopic sonography, BGH has been described as having variable echogenicity.² Computed tomography usually shows an homogeneous enhancement mass after contrast administration, but sometimes an heterogeneous and hypoattenuating pattern owing to cystic dilatation of Brunners' glands, in addition to gland proliferation can be observed.²

MRI of BGH has never been reported and shows similar findings to computed tomography. Operative or endoscopic excision is uncomplicated, and the long-term outcome is favorable.

Although they are uncommon, BGHs should be considered in the differential diagnosis of duodenal masses to avoid more aggressive surgery. Malignant transformation has rarely been reported,³ but the presence of dysplasia in our case raises the question of operative excision in all these patients and not only for symptomatic cases.²

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References 

1. Levine JA, Burgart LJ, Batts KP, et al. Brunner's gland hamartomas: clinical presentation and pathological features of 27 cases. Am J Gastroenterol. 1995;2:290–294
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2. Patel ND, Levy AD, Mehrotra AK, et al. Brunner's gland hyperplasia and hamartoma: imaging features with clinicopathologic correlation. AJR. 2006;187:715–722
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3. Brookes MJ, Manjunatha S, Allen CA, et al. Malignant potential in a Brunner's gland hamartoma. Postgrad Med J. 2003;79:416–417
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