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CASE REPORT
Year : 2016  |  Volume : 4  |  Issue : 3  |  Page : 121-124

Brunner's gland adenoma presented with duodenojejunal intussusceptions: A case report and review of literature


1 Department of General Surgery, Abu Arich General Hospital, Saudi Arabia
2 Department of Histopathology, King Fahad Central Hospital-Jizan, Saudi Arabia
3 Department of General Surgery, Rajhi University, Faculty of Medicine, Saudi Arabia

Date of Web Publication14-Nov-2016

Correspondence Address:
Tajeddinn Ibrahim Mansour
Abu Arich General Hospital, Abu Arish
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2320-3846.193988

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  Abstract 

Brunner's gland adenoma (BGA) is a very rare benign duodenal lesion, usually asymptomatic. BGA presents with duodenojejunal intussusception (DJI) is a very rare condition; only 2-3 cases have been reported. Here, we present a 45-year-old male, Yamani, presented with a 6-month history of recurrent upper abdominal pain and melena. Computed tomography (CT) with contrast revealed target sign. Laporatomy confirmed DJI with duodenal polyp that acting as a lead point. Histopathology confirmed BGA. To conclude, BGA presents with DJIs is very rare conditions; nonspecific symptoms and less dramatic presentation are possible causes of delay in diagnosis, CT with contrast is usually diagnostic.

Keywords: Brunner′s gland adenoma, duodenojejunal intussusceptions, duodenotomy


How to cite this article:
Mansour TI, Al-Dalaleh AM, Aboujoukh AJ, Shekhawat BS, Bashier OH. Brunner's gland adenoma presented with duodenojejunal intussusceptions: A case report and review of literature. Saudi Surg J 2016;4:121-4

How to cite this URL:
Mansour TI, Al-Dalaleh AM, Aboujoukh AJ, Shekhawat BS, Bashier OH. Brunner's gland adenoma presented with duodenojejunal intussusceptions: A case report and review of literature. Saudi Surg J [serial online] 2016 [cited 2021 May 14];4:121-4. Available from: https://www.saudisurgj.org/text.asp?2016/4/3/121/193988


  Introduction Top


Brunner's gland adenoma (BGA) is a rare benign proliferative duodenal lesion arising from Brunner's gland of the duodenum, accounting for 10.6% of benign tumors of the duodenum. [1] The etiology of BGA remains obscure. [1] Most of these adenomas are small, asymptomatic, and are found incidentally during endoscopy, [1] when symptomatic, they usually present with intestinal obstruction or gastrointestinal bleeding. [2] BGA presents with duodenojejunal intussusception (DJI) reported only twice. [2] BGAs are benign tumors, but malignant degeneration has been reported. [3] BGA tends to present predominantly in the fifth and sixth decades with no sex predominance.[2] Diagnosis of BGA includes ultrasound, barium meal [1] may reveal multiple small filling defect (Swiss cheese appearance in burners gland hyperplasia or smooth polypoidal lesion) [3] in case of adenoma (vacuole sign), Upper Endoscopy localizes the lesion; however, biopsies are usually negative or reveal only Brunner gland hyperplasia due to the submucosal nature of the tumor, [1],[3] a contrast-enhanced abdominal computed tomography (CT). [3] Treatment of BGA ranges from conservative in small asymptomatic lesion, [4] endoscopic snare polypectomy, and open surgical excision. [1] Here, we report a rare case of BGA present with DJI and lower GIT bleeding.


  Case Report Top


A 45-year-old male Yamani presented with a 6-month history of upper abdominal pain, increase with food intake and melena, no history of jaundice, anorexia, weight loss, dyspepsia, or diarrhea. There was no relevant family history. During this period, patient admitted several times because of severe anemia and received blood 4 times. Clinical examination, patient pale, his vitals were normal, abdomen not distended, no visible peristalsis, not tender, and no palpable masses. Standard laboratory investigations including hemoglobin, blood counts, blood sugar, and renal and hepatic function tests were all within normal limits except low hemoglobin. Abdominal ultrasonography revealed a longitudinal mass of 12 cm just below the pancreas, dilated pancreatic duct (5 mm). A contrast enhanced abdominal CT showed a "Target Sign" [Figure 1] suggestive of DJI. At laparotomy, there was an obvious DJI that easily reduced. A 2-3 cm mass was palpable at the junction of the first part (D1) and the second part (D2) of the duodenum. A duodenotomy performed along the long axis of the D1. A 2-3 cm pedunculated polyp arises from the posterior wall of the D1. Polypectomy including excision a rim of a normal duodenal mucosa. Duodenotomy closed transversely. Patient underwent smooth postoperative course. Histopathology revealed BGA [Figure 2] [Figure 3] [Figure 4]. Follow-up patient doing well. Patient return back to his work after 8 weeks.
Figure 1: Computed tomography with contrast revealed "Target Sign"

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Figure 2: Lobules of hyperplastic Brunner's glands (H and E, ×10)

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Figure 3: High-power view of Brunner's glands (H and E, ×40)

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Figure 4: Cystically dilated ducts (H and E)

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  Discussion Top


In 1688, Brunner gave a precise anatomic description of the duodenal submucosal glands and coined the term "pancreas secundarium." [1] In 1846, Middeldorpf correctly identified these glands as a separate entity, which he proposed be named Brunner's gland. [4] Salvioli reported the first adenoma of Brunner's gland in 1876. [1] Since then, 150 cases or so have been reported in literature of English language. [4] These glands are predominately found in the proximal duodenum and decrease progressively in size and number in the distal portion. [5] The main physiological function of the glands is to protect the duodenum from gastric acid by secreting both alkaline fluid and enterogastrone, an enteric hormone, which inhibits gastric acid secretion. [5] Feyrter classified the abnormal proliferation of the glands into three types: [6]

  • Diffuse nodular hyperplasia
  • Circumscribed nodular hyperplasia
  • Adenomatous hyperplasia which may be sessile or pedunculated.
In general, hyperplasia refers to lesion smaller than 1 cm, [3] whereas adenoma refers to lesion larger than 1 cm. [4] BGAs are benign, but malignant degeneration has been reported. [3] BGA tends to present predominantly in the fifth and sixth decades of man's life with no sexual predominance. [2] However, cases have been described from early infancy to 80 years of age. [5] The etiology of BGA remains obscure, [1] but association with peptic ulcer disease, Helicobacter pylori infection, chronic pancreatitis, and chronic renal insufficiency has been described. [5] However, such associations were not exist in our patient. Most of these adenomas are small and asymptomatic and are found incidentally during endoscopy. [1],[2] When BGAs are symptomatic, they usually present with intestinal obstruction or gastrointestinal bleeding. [1],[2] Levine et al. studied 27 patients with BGAs, they found bleeding complications in 40-50% of cases, of which 43% melena. [2] Obstructive symptoms are present in about 50% of patients, these include postprandial epigastric burning, discomfort, belching, bloating, nausea, and occasionally vomiting. [1],[2] In our case, there was epigastric pain related to the meal, but belching, bloating, nausea, and vomiting described previously were not exist. Severe anemia and anemic heart failure from GIT bleeding have been reported by some authors. [7] In this case, anemia is so severe that necessitate blood transfusion 4 times, but frank anemic heart failure described by some authors was not seen in our case. [7] DJI seen in this patient is very rare occurrence [1] because duodenum is fixed to the posterior abdominal wall by the peritoneum, ligament of treitz, superior mesenteric vessels, and the head of pancreas. [1] To date, only 47 cases of DJIs have been reported and usually secondary to benign duodenal tumors. [8] However, DJI secondary to BGA is a very rare condition only 2-3 cases have been reported. [2] The classical triad of abdominal pain, palpable mass, and passage of red currant jelly stools described in children is seldom seen in adult patients. However, presentations such as palpable abdominal mass, jaundice, pancreatitis, and diarrhea were reported by some authors. [1] However, these features were not seen in our patient. In this patient, the diagnosis of DJI was made based on CT abdomen with contrast that revealed the typical "Target Sign" [Figure 1], at the region of distal duodenum and proximal jejunum which is diagnostic. The sensitivity and the specificity of CT scan to diagnose intussusceptions are 71.4-87.5% and 100%, respectively. [9] There are various methods for the resection of Brunner's gland hyperplasia. For instance, endoscopic polypectomy should be performed if the lesions are <1 cm in diameter. [10] However, if the size is excessively large or if the tumor is the sessile type, then, surgical polypectomy, duodenal wedge resection, or partial gastrectomy that is extended to the duodenal bulb should be done. [10] There is one report of a duodenocephalopancreatectomy, in a Brunner's gland hyperplasia case involving the duodenal second portion with a pancreatic invasion. [10] In our case, a transduodenal polypectomy was done where the mass was excised along with a cuff of a normal duodenal mucosa.


  Conclusion Top


BGA presents with DJI and melena is a very rare condition; nonspecific symptoms and less dramatic presentation are the usual causes of delay in diagnosis. CT is usually diagnostic. The final diagnosis requires histopathology evaluation.

Acknowledgments

I would like to thank all who provided assistance during the preparation of this manuscript. Special thanks to Mohunad Osman alsaadi: A computer technician, Abo Arich General Hospital, for his great help in coordination of this manuscript. Also I would like to thank Yahia Mohammed Mahrazy an anesthetist technician who was taking many photos and videos during operations.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Gao YP, Zhu JS, Zheng WJ. Burner gland adenoma of the duodenum. A case report and literature review. World J Gastroenterol 2004;10:2616-7.  Back to cited text no. 1
    
2.
Levine JA, Burgart LJ, Batts KP, Wang KK. Brunner's gland hamartomas: Clinical presentation and pathological features of 27 cases. Am J Gastroenterol 1995;90:290-4.  Back to cited text no. 2
    
3.
Fujimaki E, Nakamura S, Sugai T, Takeda Y. Brunner's gland adenoma with a focus of p53-positive atypical glands. J Gastroenterol 2000;35:155-8.  Back to cited text no. 3
    
4.
Peetz ME, Moseley HS. Brunner's gland hyperplasia. Am Surg 1989;55:474-7.  Back to cited text no. 4
    
5.
Stolte M, Schwabe H, Prestele H. Relationship between diseases of the pancreas and hyperplasia of Brunner's glands. Virchows Arch A Pathol Anat Histol 1981;394:75-87.  Back to cited text no. 5
    
6.
Feyrter F. Brunner gland adenoma presented with duodenojejunal intussusception: A case report and review of literature 1934;293:509-26.  Back to cited text no. 6
    
7.
Khan Z. Brunner's gland adenoma: Case report with review of literature. Pak J Surg 2007;33:1-4.  Back to cited text no. 7
    
8.
Naik BM, Arjun N, Kudari A. A case of duodeno-duodenal intussusception. J Evol Med Dent Sci 2015;4:6138-42.  Back to cited text no. 8
    
9.
Barbiera F, Cusmà S, Di Giacomo D, Finazzo M, Lo Casto A, Pardo S. Adult intestinal intussusception: Comparison between CT features and surgical findings. Radiol Med 2001;102:37-42.  Back to cited text no. 9
    
10.
Iusco D, Roncoroni L, Violi V, Donadei E, Sarli L. Brunner's gland hamartoma: 'Over-treatment' of a voluminous mass simulating a malignancy of the pancreatic-duodenal area. JOP 2005;6:348-53.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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