Acute pancreatitis and upper gastrointestinal bleeding as presenting symptoms of a duodenal Brunner ’ s gland hamartoma

Departments of 1Gastroenterology and 2Internal Medicine, Bnai Zion Medical Center, The Bruce Rappaport School of Medicine, Technion, Haifa, Israel Correspondence: Dr Edy Stermer, Bnai Zion Medical Center, PO Box 4940, Haifa 31048, Israel. Telephone 972-4-8359426, fax 972-4-8359726, e-mail edystermer@yahoo.com or edy.stermer@b-zion.org.il Received for publication November 9, 2005. Accepted December 16, 2005 E Stermer, N Elias, D Keren, T Rainis, O Goldstein, A Lavy. Acute pancreatitis and upper gastrointestinal bleeding as presenting symptoms of a duodenal Brunner’s gland hamartoma. Can J Gastroenterol 2006;20(8):541-542.


CASE PRESENTATION
A 20-year-old woman was admitted to the Department of Medicine, Bnai Zion Medical Center, Haifa, Israel, for abdominal pain and melena that began three days before admission.
The pain was severe, localized to the upper abdomen, persistent and noncolicky.On admission to the medical ward, she was alert and well nourished, without scleral icterus or lymphadenopathy.She had no personal or family history of gastrointestinal disease, and there was no history of alcohol use.
On examination, the abdomen was soft but tender in the periumbilical area, without peritoneal irritation.Rectal examination showed traces of melena.Routine laboratory examinations showed a hemoglobin level of 115 g/L, a leukocyte concentration of 9.9×10 9 /L, and normal serum levels of urea, glucose, bilirubin, aspartate aminotransferase, alkaline phosphatase, electrolytes, serum cholesterol and triglycerides.The serum amylase level was 1054 U/L (normal range 30 U/L to 100 U/L) and the urinary amylase level was 5220 U/L (normal range 20 U/L to 500 U/L).The chest and abdominal x-rays were unremarkable.
The abdominal ultrasonography was normal; a subsequent computed tomography scan disclosed a slightly edematous pancreas, with normal bile ducts and liver.A large (5 cm) filling defect with soft borders was revealed in the second part of the duodenum, partially obstructing the lumen (Figure 1).Gastroscopy showed a 7 cm long, lobulated mass with an ulcerated surface, freely moving on a long pedicle in the proximity of the papilla Vateri (Figure 2).
Treatment with omeprazole and analgesics resulted in the resolution of abdominal pain four days after admission.The polypoid mass was dragged into the stomach, and a one-stage endoscopic polypectomy was performed using 40 W of coagulation current.Within five days, the amylase levels progressively decreased to normal.The hemoglobin levels remained stable.On histological examination, the polyp was formed by nondysplastic, lobulated Brunner's glands, with intervening bands of fibrous tissue, adipose cells and lymphoid cells (Figure 3).
On follow-up visits three and seven months after discharge, the patient remained asymptomatic.Her most recent hemoglobin level was 127 g/L and the diastase level was normal.A fecal occult blood test examination was negative.

DISCUSSION
Duodenal tumours are infrequent findings, while Brunner's cell tumours are even less frequently diagnosed.Brunner's glands are found at the gastrointestinal junction and extend for variable distances distally in the wall of the proximal small intestine.The secretory units of Brunner's glands consist of epithelial tubules that show frequent distal branchings.They produce mucin glycoproteins, limited amounts of bicarbonate, epidermal growth factor, bactericidal factors and proteinase inhibitors.All these products contribute to the mucosal surface protection.Brunner's gland hamartomas were first described by Cruveilhier in 1835 (1) in a patient with fatal duodenal intussusception.These tumours are usually located in the duodenal bulb or in the second duodenal portion.They are benign small bowel tumours, with fewer than 150 cases reported in the English literature until the end of the last century (2); they represented 13% of a large, Australian series of benign small bowel tumours (3).Brunner's gland hamartomas are more commonly discovered during an episode of acute upper gastrointestinal bleeding (4)(5)(6), in evaluating the cause of iron deficiency anemia (7) or because of gastrointestinal obstructive symptoms (8,9).Unusual among the presentations of Brunner's gland hamartoma is a biliary fistula (10) as an imitator of carcinoma of the head of pancreas (11) or causing pancreatitis (12).The presenting symptom in our patient was acute upper gastrointestinal bleeding, which is a common presentation of this tumour.However, she also had severe abdominal pain and hyperamylasemia, consistent with acute pancreatitis.The association of upper intestinal bleeding and hyperamylasemia in the presence of a juxtapapillary mass is suggestive of obstruction of the papilla Vateri by the duodenal mass.The remission of symptoms after ablation of the duodenal tumour and the return to normal of the laboratory abnormalities may attest that the intestinal bleeding and pancreatitis were both caused by the duodenal mass.Furthermore, the patient has remained completely asymptomatic after seven months of follow-up.There was no ongoing anemia; her most recent hemoglobin level was 127 g/L and the diastase level was normal.In reviewing the literature, we could not find any other report of Brunner's cell duodenal hamartoma presenting with both intestinal bleeding and pancreatitis.

Figure 1 )Figure 2 )
Figure 1) Abdominal computed tomography scan showing the large duodenal polyp