Brunner Gland Hamartomas—Uncommon Presentations and Endoscopic Management

Abstract

Brunner gland hamartoma (BGH) is a rare condition that requires a high clinical suspicion to diagnose. Large hamartomas may initially present with iron deficiency anemia (IDA) or symptoms suggesting intestinal obstruction. Barium swallow may demonstrate the lesion, but endoscopic evaluation is the acceptable first line management unless a concern for underlying malignancy. The present case report and literature review highlight the uncommon presentations and endoscopic role in large BGHs management. Internists should consider BGH in their differential, especially in patient with occult bleeding, IDA, or obstruction, which can be treated with endoscopic resection of large sized tumors by trained experts.

Introduction

Brunner gland hamartoma (BGH) is rare benign tumor of the duodenum that requires a high clinical suspicion to diagnose. Although it may be discovered incidentally, it can be the cause of the underlying gastrointestinal bleeding (GIB) or intestinal obstruction. Here, we present a case of anemia and vomiting due to BGH followed by a literature review of the current practice of diagnosis and management of large BGH.

Case Report

A 66-year-old man with a history of hypertension, type 2 diabetes, and vitamin B12 deficiency was referred to the gastroenterology clinic for iron deficiency anemia (IDA), intermittent non-bloody bilious vomiting, and abdominal discomfort. He also had rare occurrences of hematochezia. He reported no early satiety, unintentional weight loss, or change in bowel habits. Family history is significant for colon cancer in his father at 59-years-old. Physical exam was significant for pallor and abdominal exam was soft and nontender. Laboratory workup was significant for normocytic anemia with hemoglobin of 9.6 g/dL and iron deficiency with iron level of 7 mcg/dL, ferritin of 3.5 ng/mL, transferrin saturation of 7%, and total iron binding capacity 411 mcg/dL. Intrinsic factor antibody, folate, thyroid stimulating hormone, and comprehensive metabolic profile were normal. Patient underwent esophagogastroduodenoscopy (EGD) that revealed a 2-cm hiatal hernia, grade B esophagitis, and a large 50-mm partially obstructing pedunculated duodenal polyp extending from the duodenal sweep to the second portion of the duodenum (Figure 1). Due to the thickness of the stalk, polypectomy was difficult with the standard polypectomy program. Rather, an endoloop was first placed, and hot snare polypectomy was then performed by increasing the maximum Watts used. Histopathological evaluation of the polyp showed benign Brunner’s gland hyperplasia (Figure 2). Colonoscopy showed low-risk adenomas, arteriovenous malformations (AVMs) that were managed by argon plasma coagulation (APC), diverticulosis, and nonbleeding hemorrhoids. The patient’s IDA and abdominal symptoms have significantly improved following resection of the duodenal polyp with no further vomiting or intermittent abdominal discomfort. The IDA was deemed to be due to duodenal BGH.

Figure 1.

Esophagogastroduodenoscopy demonstrating the pedunculated duodenal polyp.

Figure 2.

Histological evaluation of the pedunculated duodenal polyp. Polypoid duodenal mucosa showing Brunner’s gland hyperplasia and chronic inflammation, focal villous blunting noted, with no evidence of increased intraepithelial lymphocytes, adenomatous changes, or dysplasia.

Discussion

Hamartomas are benign tumors that are fundamentally comprised of disordered replications of normal tissue cells characterized by an abnormal cellular mixture (eg, smooth muscles, adipose tissue, and cartilage). ¹ Brunner gland hamartoma (BGH) is an uncommon benign gastrointestinal (GI) tumor accounting for 5% to 10% of benign duodenal tumors with an incidence rate <0.01%.^2,3 Brunner glands produce anti-acid secretion that coat the duodenal epithelium protecting it from stomach acidity. Thus, it was postulated that BGH are commonly seen in patients with hyperchlorhydria and/or Helicobacter pylori infection.^4,5 However, such hypothesis has been controversial. ⁶

The most common symptoms of BGHs are abdominal pain and melena, occurring in just over 50% of patients in a meta-analysis. ⁷ The hamartoma size has also been correlated to the symptoms. Small BGHs have an average size of 1.6 cm and are commonly asymptomatic. Large sized hamartomas (>2 cm) are likely to present with gastrointestinal bleeding (GIB), symptomatic anemia, or obstructive symptoms. ⁸ The location of BGH may correlate with the severity of symptoms. BGHs in the first portion of the duodenum usually present with less severe symptoms; whereas, distal lesions manifest more profoundly, such as with obstructive symptoms since the small bowel becomes narrower distally. Similarly, our patient presented with symptoms of partial obstruction including vomiting as well symptomatic IDA secondary to the BGH located in the second part of the duodenum.

Imaging studies, such as an upper GI series or computed tomography (CT) scan, can detect the BGH location. Once discovered by imaging, EGD can diagnose and potentially treat the condition. Histologic evaluation usually demonstrates submucosal hyperplasia of the gastric mucosa consistent with BGH. ⁹ Management of BGH is either endoscopic or surgical and is usually based on the tumor size and malignancy potential. The size of the lesions resected endoscopically usually ranges from 0.5 to 5 cm. ¹⁰ However, surgery is reserved for lesions that are large, endoscopically unresectable, or have a suspicion for malignancy. ¹¹ Although there is a high removal success rate, about 37% of polyps recur within an average of 26 months, commonly in polyps less than 2 cm. ¹² Thus, postresection surveillance is recommended for early detection of recurrence.

In summary, Brunner gland hamartomas are an extremely rare benign hyperplastic lesions of the GI tract and are most commonly incidentally detected. Hence, we suggest that clinicians should be cognizant of considering BGH in their differential, especially in case of obstruction, occult GI bleeding, and IDA.