A 44-year-old Chinese man with no previous comorbidities, no chronic medications, and no surgical history presented to the emergency department with an episode of self-limiting acute epigastric pain, nausea, and vomiting for 2 days.1-3 His bloating was worse after meals and increased in relation to the portion of food consumed. He reported long-standing intermittent epigastric discomfort with bloating for 3 months. His vital signs were within normal limits. The physical examination revealed no remarkable findings. His abdomen was soft without any palpable abdominal mass, guarding, or rebound tenderness. Laboratory investigations did not reveal any significant abnormality.
Upper gastrointestinal endoscopic examination performed showed large amount of food residue in the stomach, although the patient was adequately fasted. His stomach was capacious, with a distorted antrum. The first part of the duodenum was distorted and dilated with prominent valvulae conniventes (Figure 1), which are normally only seen starting in the distal length of the duodenum closer to the duodenojejunal flexure.4 Computed tomography showed a long segment duodenojejunal intussusception, seen as a target-like mass, with proximal dilatation of the stomach (Figure 2). Subsequent surgical laparotomy revealed a large polyp measuring 9.0 × 5.0 × 4.0 cm, arising from the first part of the duodenum (Figure 3). Histology showed features of a benign Brunner's gland adenoma (BGA) consisting of multiple nodules of closely packed Brunner's glands, separated by fibromuscular septa. No mitotic figures were observed (Figure 4). Our patient underwent an uneventful postoperative recovery with resolution of symptoms.
Brunner's glands are named after the Swiss anatomist Johann Conrad Brunner, who first described them in 1,688.5 Hyperplasia of Brunner's glands to form a lesion greater than 1 cm is described as a Brunner's gland adenoma (BGA) or Brunner's gland hamartoma.6 Brunner's glands are located predominantly in the first part of the duodenum and produce alkaline secretions containing mucus, pepsinogen, and urogastrone in response to acid stimuli. Their size and number decrease progressively in the distal parts of the duodenum. Most BGAs are in the duodenal bulb (57%), followed by second (27%) and three times a day (7%) parts of the duodenum. They are seldom found in the pyloric canal (5%), jejunum (2%), or proximal ileum (2%).6
The etiology and pathogenesis of BGAs are not fully understood. Suggested causes include factors causing chronic local irritation, such as Helicobacter pylori infection, Billroth II reconstruction, or chronic pancreatitis.7,8
BGA is a benign lesion, but there have been case reports describing neoplastic lesions arising from the Brunner's gland.9 Most patients with BGA are asymptomatic and are only incidental during endoscopy or imaging studies. Symptomatic patients with BGA tend to have adenomas larger than 2 cm in size and commonly present with gastrointestinal bleeding or obstructive symptoms.10 Less common presenting symptoms include intussusception, biliary fistula, recurrent pancreatitis, or obstructive jaundice. To date, there have been few published case reports of BGA associated with acute or chronic gastric outlet obstruction and intussusception.
The diagnosis of BGA can be challenging. Imaging findings are usually nonspecific and may suggest adenoma, adenocarcinomas, lymphoma, carcinoid tumors, gastrointestinal stromal tumors, leiomyomas, leiomyosarcomas, and metastatic disease.11 Being submucosal in origin, traditional endoscopic punch biopsies are usually negative. Because a Brunner's gland may be covered by normal mucosa, confirmatory diagnosis is only established after a deep endoscopic or a surgical biopsy, with consequent histological examination of the excised mass.12 Treatment options include endoscopic removal of small and/or pedunculated stalked lesion, endoscopic mucosal resection with piecemeal technique for larger adenomas, and surgical resection where endoscopic therapy is not feasible.13 Endoscopic management is the preferred initial mode of therapy. Where feasible, endoscopic ultrasound can provide further information about the depth of involvement and any submucosal vasculature that may be associated with the BGA. Cases of BGA with an intact muscularis propria layer can be managed endoscopically with endoscopic submucosal dissection.14 In our case, surgery was preferred because of a large lesion complicated with intussusception.
Author contributions: R. Teh: manuscript writing, data acquisition, approval of final version, and agreement to all aspects of work. TW De, C. Weihao, and AA Qurishi: manuscript review, data acquisition, approval of final version, and agreement to all aspects of work. AA Qurishi is the article guarantor.
Financial disclosure: None to report.
Informed consent was obtained for this case report.
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