Archives of the Balkan Medical UnionCopyright © 2017 Balkan Medical Union

vol. 52, no. 4, pp. 453-457December 2017

RÉSUMÉ

Approche endoscopique d’un cas rare d’obstruction d’un hamartome geant duodenal de la glande de Brunner

IntroductionLes hamartomes des glandes de Brunner sont des lésions très rares, petites et bénignes, fréquemment situées dans l’ampoule du duodénum. En géné-ral, accidentellement trouvées lors d’une œsopha-go-gastro-duodénoscopie de routine, ces lésions sont asymptomatiques, les patients peuvent présenter des symptômes d’obstruction duodénale ou d’hémorragie secondaire à l’ulcération. L’excision chirurgicale est requise surtout si la lésion a de grandes dimensions. L’examen pathologique est d’une importance majeure pour la confirmation du diagnostic.Présentation de cas cliniqueNous rapportons ici un cas d’un patient de 64 ans qui s’était présenté au service d’urgence pour des vomisse-ments d’aliments partiellement digérés, sans hémor-ragie. L’endoscopie digestive supérieure a révélé une masse polypoïde de 5/4 cm, avec un pédicule de 1 cm, situé dans l’ampoule duodénale. Le polype a été

ABSTRACT

IntroductionBrunner’s gland hamartomas are very uncommon. They are small, benign lesions, frequently located in the bulb of duodenum. Usually found incidentally dur-ing routine esophago-gastro-duodenoscopies, these le-sions are frequently asymptomatic, but some patients may present with symptoms of duodenal obstruction or hemorrhage secondary to ulceration. Surgical exci-sion is required, especially if the lesion has large di-mensions. Pathological examination is the most impor-tant in confirming the diagnosis.Case presentationWe report a case of a 64-year-old female patient, who presented to the emergency room for vomiting partial-ly digested food, without blood. Upper digestive endos-copy revealed a polypoid mass, with a 1 cm stalk, lo-cated in the duodenal bulb. The polyp was successfully endoscopically resected and the pathological diagnosis was of Brunner’s gland hamartoma. Histologically, this polyp consisted of the components of Brunner’s gland cells, as well as glandular, adipose and muscle cells.Conclusions

CASE REPORT

ENDOSCOPIC MANAGEMENT OF A RARE CASE OF OBSTRUCTIVE GIANT DUODENAL BRUNNER’S GLAND HAMARTOMA

Gelu C. Rosianu1,2, Anca Evsei1,3, Mircea Gheorghe4, Adelina Birceanu–Corobea3, Narcis Copca4

1 CESITO Center, „Sfanta Maria“ Clinical Hospital, Bucharest, Romania2 Department of Gastroenterology, „Sfanta Maria“ Clinical Hospital, Bucharest, Romania3 Department of Pathology, „Sfanta Maria“ Clinical Hospital, Bucharest, Romania4 Department of Surgery, „Sfanta Maria“ Clinical Hospital, Bucharest, Romania

Corresponding author: Rosianu Cristian

CESITO Center, „Sfanta Maria“ Clinical Hospital, Bucharest, Romania

e-mail: Rosianu_Cristian@yahoo.com

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INTRODUCTION

Brunner’s gland hamartoma is a rare tumor, a nodular hyperplasia of the normal Brunner’s gland with an unusual mixture of normal tissues, including ducts, adipose tissue and lymphoid tissue. It repre-sents 10.6% of duodenal benign tumor1.

Brunner’s gland hamartomas are typically found in the duodenal bulb and in the second portion of the duodenum, proximal to the sphincter of Oddi2.

Their main physiological function consists in producing a mucus-rich, bicarbonate-containing alkaline secretion that, among other features, neu-tralizes the acidic content of chime and gastric acid3. Although proliferative lesions of Brunner’s gland do not have yet an established nomenclature, and dis-tinction between various lesions is arbitrary, it is im-portant to identify this pathology as it is benign, be-cause there are no documented cases of malignancy4.

Patients with Brunner’s gland hamartoma may be asymptomatic and the lesion is usually discovered incidentally5,6. Among possible symptoms and com-plications caused by Brunner’s gland hamartoma, duodenal obstruction, intussusception, obstructive jaundice, pancreatitis and bleeding are included. At upper digestive endoscopy, Brunner’s gland hamar-toma is a submucosal mass that often has a stalk, which points out that an endoscopic mucosal biopsy may often be inconclusive as the biopsy device does not penetrate the submucosa7. Macroscopically, this lesion is usually pedunculated, polypoid and micro-scopically it iscomposed of normal Brunner glands combined with variable amounts of adipose, smooth

muscle, and lymphoid tissue, as well as with sclerotic areas of Brunner glands in some cases.

Treatment of Brunner’s gland hamartoma consists of endoscopic polypectomy, which is more cost-effective and less invasive than surgery. The med-ical decision usually depends on location and size of the tumor, as well as on the patient’s symptoms and status8,9.

This article reports a case of giant Brunner’s gland hamartoma, and reviews brief ly its clinical presentations, pathological features and therapy.

CASE PRESENTATION

A 64 year-old male patient was admitted to our hospital for epigastric pain and vomiting, symptoma-tology that began about a month before, progressing towards a worse clinical status.

The physical examination revealed epigastric pain on palpation. Laboratory investigations revealed mild hypochromic and microcytic anemia.

Abdominal ultrasound performed in the emer-gency room showed the stomach filled with food debris, raising the suspicion of gastric stasis. Upper digestive endoscopy was performed and gastric sta-sis, along with a stalked giant polyp located on the posterior wall of the duodenum, near the junction of its first and second portions, were revealed. The surface of the tumor was smooth, with mild erosions and ulcers. Multiple biopsy specimens were taken and interpreted as “mucosal mild-medium atypia“. Tumor markers CA 19.9 and CEA were within normal range.

réséqué avec succès par endoscopie et le diagnostic pathologique était celui d’un hamartome de la glande de Brunner. A l’examen anatomo-pathologique, ce pol-ype était composé de cellules de la glande de Brunner, ainsi que de cellules glandulaires, adipeuses et muscu-laires.ConclusionsLes hamartomes de la glande de Brunner sont des tumeurs duodénales rares, survenant chez des patients d’âge moyen, présentant une hémorragie gastro-intes-tinale, des symptômes obstructifs ou des découvertes accidentelles. L’excision chirurgicale ou endoscopique n’est pas compliquée et le résultat à long terme est fa-vorable. La particularité de notre cas est donnée par le fait que cette lésion était très volumineuse, elle a provoqué des symptômes comme les vomissements et a été réséquée uniquement par endoscopique.

Mots-clés: hamartome, glande de Brunner, exérèse endoscopique.

Brunner’s gland hamartomas are rare duodenal tu-mors occurring in middle-aged patients that present either with gastrointestinal hemorrhage, obstructive symptoms, or as an incidental finding. Surgical or en-doscopic excision is uncomplicated, and the long-term outcome is favorable. The particularity of our case is given by the fact that this lesion was very large, it caused symptoms such as vomiting and was managed solely by endoscopic resection.

Key words: giant hamartoma, Brunner’s gland, endo-scopic resection.

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Endoscopic resection of the polyp was suggested to the patient and after careful consideration, togeth-er with the surgical team, endoscopic polypectomy was chosen as the first option. The resection was pre-ceded by submucosal injection of diluted 5 ml polygel with epinephrine 1:10,000 solution into the base of the polyp, followed by efficaciously removed polyp using electrosurgical snare. Diffuse bleeding occurred immediately after polypectomy, but efficient endo-scopic hemostasis was performed by using hemoclips.

The resected specimen showed a lobulated, poly-poid mass, measuring 5 cm × 4 cm, projecting into the duodenum. The stalk measured 1 cm in diameter and 1 cm in length (Figure 1,2).

The site of resection was closed with hemoclips (Figure 3) to prevent post-polypectomy bleeding and perforation.

After 48 hours post-polypectomy, the patient was allowed to resume feeding. On follow-up, there was no rebleeding and no residual lesion.

Pathological evaluation of Brunner’s gland hamartoma revealed: on gross examination, the tu-mor showed a light brown colored, firm, peduncu-lated, polypoid mass, measuring 5x4x2.5 cm. The surface was smooth, with erosions and ulceration on the surface. The cut surface was firm, yellowish and lobulated, with a few slit-like spaces. On microscopic examination, the tumor was covered by small intesti-nal mucosa and composed of lobules of proliferated Brunner glands with adipose tissue metaplasia; in addition, there was moderate infiltration with lym-phocytes, with a few lymphoid follicles. Some of the ducts were cystically dilated. The covering mucosa was flattened and showed hyperemia, foci of hem-orrhage and mild infiltration of lymphocytes and plasma cells. The hamartomatous components were not present in the stalk of these histological findings. Finally, a diagnosis of Brunner’s gland hamartoma was made. (Figures 4-9)

DISCUSSION

Brunner’s gland hamartoma has a rather un-clear etiology. The average size is between 0.5 cm and 12 cm. The most frequent location is on the posterior wall of the duodenum, at the junction be-tween its first and the second part. In rare cases, it

Figure 3. Endoscopic images after polypectomy.

Figure 1,2. Endoscopic images of Brunner’s gland hamartoma.

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Figure 4. (H&E 50x) Tumor covered by small intestinal mucosa (thin blue arrow) and composed of lobules

of proliferated Brunner’s glands (large thick arrow) separated by irregular bands.

Figure 6. (H& E 50x) Lobules of proliferated Brunner’s gland with fibrous bands and adipose tissue metaplasia

Figure 8. (H&E 200) Covering mucosa showing hyper-emia and mild infiltration with lymphocytes

Figure 5. (H& E 50x) Lobules of proliferated Brunner’s glands separated by irregular bands of fibromuscular

stroma with moderate infiltration by lymphocytes.

Figure 7. (H& E 50x) Lobules of proliferated Brunner’s glands with ducts separated by irregular

bands of fibromuscular stroma and lymphocytes with a few lymphoid follicles

Figure 9. (H&E 200) Brunner’s glands with dilated ducts and fibrous bands

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can be located in the second and third part of the duodenum. Brunner’s gland hamartomas are usually asymptomatic, benign incidental findings, discovered when performing upper gastric endoscopy or barium swallow10.

The most common symptoms are upper diges-tive hemorrhage and emesis. In the case of a giant Brunner’s gland hamartoma, the upper digestive hemorrhage may be exteriorized as hematemesis or melena; occasionally, this bleeding may be massive and very rarely fatal11.

According to the endoscopic classification, these hamartomas can be sessile, pedunculated, polypoid tumor masses or submucosal tumors12.

Differential diagnosis should be done with: ma-lignant tumors, adenomatous polyps, ectopic pan-creas, leiomyoma, GIST, neuroendocrine tumors, ampulla of Vater and pancreatic neoplasm with duo-denal invasion. The treatment for Brunner’s gland hamartomas is recommended in symptomatic cases or to prevent complications (such as bleeding, ob-struction)13.

It is still controversial whether asymptomatic Brunner’s gland hamartoma incidentally discovered needs surgical removal14. Endoscopic resection is in-dicated for stalked polyps or for confirming the be-nign character of the lesion. Surgical treatment is pro-posed when there is a pathological proven malignant component, submucosal development or for those le-sions difficult to resect by endoscopy. Brunner’s gland hamartomas are benign, but there are documented cases in the literature with dysplasia found in the cov-ering intestinal mucosa. There have been no reports of malignant Brunner’s gland hamartomas.

CONCLUSIONS

Brunner’s gland hamartoma is a rare duodenal tumor. Symptomatology is present in large duodenal tumors. An alternative to surgical resection is the en-doscopic resection; pre-resection histological diagno-sis is not always easy.

There are no reported recurrences follow-ing Brunner’s gland hamartoma removal and the long-term prognosis is excellent.

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