Brunner's gland hyperplasia presenting with melena: A case report

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Case Report

Brunner’s gland hyperplasia presenting with melena: A case report

Saâd Rifki Jai

^*

, Driss Erguibi, Rachid Boufettal, Driss Khaiz, Farid Chehab

Service de Chirurgie Viscérale III, CHU Ibn Rochd, Casablanca, Morocco

a r t i c l e i n f o

Keywords:

Melena Brunner’s gland Hamartoma

a b s t r a c t

:

Polypectomy

Introduction

Brunner’s gland adenoma is a rare duodenal benign tumour, usually located in the duodenal bulb. It is often non-symptomatizing but can occasionally cause digestive haemorrhage or duodenal obstruction[1–3], requiring surgical or endoscopic excision of the lesion[4–6].

We report on a female patient admitted to the surgical department for lithiasis of the gall bladder, having a history of chronic anaemia which necessitated blood transfusion, and during admission, she presented with digestive haemorrhage in the form of melena unveiling a polypoid hyperplasia of Brunner’s glands. Through this case presentation and a review of the literature, we provide prognostic aspects of this type of duodenal tumour.

Case report

Mrs. G.A, aged 46 years, with a history of microcytic anaemia for 19 months, treated with repeated blood transfusions over the preceding 2 months was then admitted to the surgical department in March 2005 suffering from right hypochondrial and epigastric pain progressing over the preceding 4 months.

The clinical examination revealed that the patient was in good general condition, no jaundice, blood pressure (BP) was 100/

70 mm hg and BMI was 28.57 kg/m². The haemoglobin level (Hb) at admission was 13.4 g/dl, alkaline phosphatase 123 U/L, gamma glutamyl transferase 35 U/L, transaminases 20 U/L. Abdominal ultrasound revealed gall bladder stones with a normal wall, while the common bile duct (CBD) was dilated (10 mm) with a tapering distal end suspicious for a stricture. Due to epigastric pain, upper gastrointestinal endoscopy was done, biopsies from the gastric

mucosa revealed atrophic pangastritis and minimally active chronic gastritis of the fundus with presence ofHelicobacter pylori (HP).

Accordingly, triple therapy forH. pylorieradication was admin- istered. Two days later, the patient presented with signiﬁcant melena with a haemoglobin level of 6 gm/dl and a haematocrit level of 17.5%, Urgent blood transfusion of 3 units of packed red blood cells was done.

A second endoscopy showed petechial diffuse erythematous gastritis with the presence of ulcerated polypoid formations in the duodenum. These were purplish-blue, prevailing in the proximal part of the 2nd part of duodenum (DII) and extending into the third part of duodenum (DIII). It was easily bleeding on contact and therefore difﬁcult to biopsy. Such lesions were suggested to ac- count for the sonographic dilatation of the CBD through papillary inﬂammation or compression. Abdominal CT detected a tumoural process with tissue density, extending from DII to DIII, 5445 mm in diameter, hypodense that moderately enhanced after injection of contrast. This process extended to the peri-duodenal jejunal mesenteric fat, encircling the mesenteric pedicle and reaching the inferior vena cava. The liver was normal.

Such findings suggested a mesenchymal tumour (Fig. 1). A lap- arotomy for diagnostic and therapeutic purposes was carried out and revealed an inflammatory duodenal wall. Transpyloric duodenotomy allowed access to the tumour (Fig. 2). A soft pedunculated purplish-blue mass measuring 7 cm (longest axis) with a smooth surface could be seen. The base measured 15 mm, seated on the posteroexternal face of the first part of duodenum and prolapsed in the duodenal lumen to DII. Peribasal excision of the tumour at 4 mm from its root with pyloroplasty was conducted. The gall bladder showed stones and had a thin wall.

Cholecystectomy was conducted. No complications ensued post-operatively with no recurrence of haemorrhage. Post-operative haemoglobin was 9 g/dl. The histopathologic study of the operative part, weighing 40 g and measuring 734 cm, showed

doi:10.1016/j.ajg.2010.07.010

* Corresponding author. Tel.: +212 661203848.

E-mail address:saadjai@yahoo.fr(S.R. Jai).

Arab Journal of Gastroenterology 11 (2010) 171–173

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a nodular proliferation made of islets of normal Brunner’s glands, separated by vascular walls without evidence of dysplasia or abnormal mitosis.

The diagnosis of a polypoid hyperplasia of Brunner’s glands without malignant signs was established (Fig. 3). The clinical evo- lution was favourable without bleeding recurrence within a fol- low-up period of 24 months.

Discussion

Brunner’s glands are tubulous ramified mucoid glands gathered in lobules of 0.5–1 mm in diameter separated by finely fibrous sep- ta. Such glands are located in the deep part of the mucosa and in the submucosa of the pylorus and in the proximal part of the duodenum. They secrete alkaline mucous and bicarbonate that covers the duodenal mucosa and protects against the acidity of gastric secretions[4,5].

In 1688, Brunner provided the anatomic description of these glands beneath the duodenal mucous naming these ‘pancreas secundarium’. In 1846, Middledurpf identiﬁed these glands as a whole entity and proposed the term ‘Brunner glands’. Salvioli reported the ﬁrst case of Brunner‘s gland adenoma in 1876.

Since ﬁrst described, only 150 cases have been reported in the literature[4,6]. The proper naming of the proliferation of these Brunner’s glands remains controversial, with suggestions of deno- ma, hamartoma and polypoid hyperplasia. In general, it is a rare benign tumour of the duodenum, representing 5–10% of the duodenal benign tumours and less than 1% of the small intestinal tumours. It affects adults between 40 and 60 years without predominance in either sex. This tumour is often asymptomatic, accidentally discovery during endoscopy, mainly in suspected cases of gastroduodenal ulcer or gastroesophageal reﬂux disease [1,7,8]. On the other hand, it can present with upper gastrointestinal haemorrhage, whether haematemesis and/or melena, or chronic anaemia[2,3]secondary to ulceration of the mucosa cover- ing the tumour. In some cases, it can cause duodenal obstruction with abdominal pain and vomiting, episodes of pancreatitis with obstruction of the bile ductor exceptionally by acute intestinal invagination[5]. In our case, the patient had history of chronic anaemia treated by blood transfusion during her admission to hos- pital for gall bladder stones and presented with melena revealing polypoid hyperplasia of Brunner glands.

Endoscopy with biopsy is the goldstandard for the diagnosis of these lesions[1,5]. The lesion is usually seated in the duodenal bulb (in 57% of the cases), but can extend to the second (27%) and third (5%) parts of the duodenum or to the proximal part of the jejunum (2%)[2]. Park et al. described the ﬁrst case of the localisation in the 4th part of duodenum and underlined the difﬁculty of detection of this lesion through standard upper gastrointestinal endoscopy and the usefulness of angiography and abdominal scan[2].

Macroscopically, the size of the lesion varies from 0.5 to 12 cm [4,5]most often between 1 and 6 cm[6,9]. The typical endoscopic aspect of this tumour is that of a single pedunculated mass with a smooth (in 88% of cases) or sessile (in 11% of cases) surface [6,10]; it can sometimes be ulcerated, accounting for digestive haemorrhage.

Endoscopic ultrasound is useful in the diagnosis of Brunner’s gland hyperplasia, in which the lesion appears as a heterogeneous hypoechoic area, involving some cystic areas involving the ﬁrst till third endosonographic wall layer structure, corresponding to mucosa and the duodenal submucosa respectively[6,11].

No characteristic CT description has been provided in the literature[9]; however, the lesion frequently appears as a duodenal process containing some cystic images and fatty zones enhanced after contrast injection[3].

The pre-operative histologic diagnosis of Brunner’s gland hyperplasia is not always easy, and endoscopic biopsies are rarely conclusive. This mainly occurs if the biopsy has not been deeply ta- ken to reach the submucosa because the tumour is entirely covered with normal duodenal mucosa[4].

Histologically, the lesion is well deﬁned and non-capsulated, made of proliferation of normal Brunner’s gland isles while pre- serving lobular architecture separated by ﬁbro-muscular stroma.

Fig. 1.Abdominal computed tomographic scan with intravenous contrast: tumoural mass of DII extending into DII measuring 5445 mm in diameter with enhancement of lesion after IV contrast.

Fig. 2.Operative ﬁnding of a polypoid mass with a smooth surface, after duodenotomy.

Fig. 3.Histological ﬁnding showing a Brunner’s gland polypoid hyperplasia (Haematoxylin and Eosin40).

172 S.R. Jai et al. / Arab Journal of Gastroenterology 11 (2010) 171–173

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Sometimes the lesion contains cystic dilatations of acini and glandular structures. There is no cellular atypia nor mitosis, and the musculosa is always intact[3,5,8,10].

The review of the literature shows a considerable variation in the designation of these tumours, with opinion differing between adenoma, hyperplasia and hamartoma[5]. The eventual classifica- tion of the WHO describes hyperplasia of Brunner’s glands by an abnormal increase in their number without other morphologic modification, and the term hamartoma can be applied to certain lesions made of a mixture of Brunner glands, adipose and lymphoid tissue. Some authors have used the term Brunneroma, indicating hyperplasia or adenoma [5,4]. In our case, the histopathologic examination revealed a proliferation of Brunner’s glands without morphologic modification, and so it is a polypoid hyperplasia rather than Brunner’s gland hamartoma.

Brunner’s hamartomas are considered in the majority of cases reported in the literature as benign tumours with no malignant potential. Most authors suggest conservative treatment in asymptomatic patients[4,8,10]. Black et al. showed in his case the long interval (more than 20 years) between the diagnosis and the appearance of clinical signs in a 65-year-old patient, conﬁrming the benign nature of this entity[9]. Nevertheless, review of the literature reveals three cases of hamartomas presenting foci of dysplasia, with cellular atypia and mitosis[5,10,12]. These data push us to ask whether a malignant transformation of these tumours – which have long been considered entirely benign – is possible. This would entail therapeutic, surgical or endoscopic resection even in asymptomatic cases.

So far treatment remains controversial in asymptomatic patients. Certain authors have proposed conservative management with surveillance, considering the Brunner hamartoma an exclu- sively benign tumour[4,8]; others have preferred endoscopic excision in order to avoid haemorrhagic complications that could be fatal. In symptomatizing patients, the treatment is always endoscopic or surgical tumour resection after duodenotomy[4–6]. Sur- gical resection is advised in case of sessile lesions, in case of diameters greater than 2 or 5 cm, or in some cases of clinical emer- gency. Endoscopic excision is preferred in the case of small size or pedunculated lesions[4–6].

Short-term and long-term prognosis of Brunner Hamartomas is excellent, and no cases of recurrence have been reported in the literature[4,6].

In conclusion, digestive haemorrhage can be the ﬁrst manifesta- tion of Brunner’s hamartoma. It is rarely massive, necessitating immediate surgical or endoscopic intervention.

Conﬂicts of interest

The authors declared that there was no conﬂict of interest.

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