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CASE REPORT
Year : 2016  |  Volume : 35  |  Issue : 4  |  Page : 456-459

Bouveret's syndrome: a report of two cases


Gastroenterology Surgical Center, Mansoura University, Mansoura, Egypt

Date of Submission03-Mar-2016
Date of Acceptance15-Apr-2016
Date of Web Publication28-Nov-2016

Correspondence Address:
Mohamed El Sorogy
Gastroenterology Surgical Center, Mansoura University, Gehan Street, Mansoura 35516
Egypt
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1110-1121.194730

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  Abstract 


Introduction Bouveret's syndrome, a rare and clinically challenging condition, is a variant of gall stone ileus caused by impaction of a large gallbladder stone in the duodenum secondary to a cholecystoduodenal fistula.
Case presentation We present two cases of duodenal obstruction in old patients due to gall stone migration and impaction in the duodenum. Both were treated surgically.
Discussion Bouveret's syndrome is a rare variant of gall stone ileus. Risk factors include old age, female sex, large-sized stones, and long history of gall stone disease. Treatment options include extracorporeal shock wave lithotripsy, endoscopy, and surgery.
Conclusion Bouveret's syndrome is a clinically challenging diagnosis to make. Although surgical stone extraction remains the classic treatment option with the least failure rates, leaving the fistula alone is associated with the least morbidity and mortality rates.

Keywords: Bouveret, gall stone, ileus


How to cite this article:
El Sorogy M, Hanafy EE, Hamed H, Atef E, Rafee AA, El-Ebeidy G. Bouveret's syndrome: a report of two cases. Egypt J Surg 2016;35:456-9

How to cite this URL:
El Sorogy M, Hanafy EE, Hamed H, Atef E, Rafee AA, El-Ebeidy G. Bouveret's syndrome: a report of two cases. Egypt J Surg [serial online] 2016 [cited 2020 Jun 4];35:456-9. Available from: http://www.ejs.eg.net/text.asp?2016/35/4/456/194730




  Introduction Top


Bouveret's syndrome, a rare and a clinically challenging condition, is a variant of gall stone ileus caused by impaction of a large gall bladder stone in the duodenum secondary to a cholecystoduodenal fistula [1]. With gall stone ileus itself being a rare cause of intestinal obstruction accounting for less than 4% of the cases, Bouveret's syndrome represents 1–4% of gall stone ileus cases, with up to 300 cases reported in the literature until 2008 [2].

We present two cases of Bouveret's syndrome with stones impacted in the third part of the duodenum managed successfully surgically.


  Case reports Top


Case 1

A 72-year-old diabetic man was referred to our outpatient clinic with 2-week history of persistent vomiting, recurrent abdominal pain, weight loss, and absolute constipation. History of recurrent attacks of biliary colics was irrelevant. His previous surgical history included umbilical hernia repair 40 years ago. The abdomen was not distended but there was epigastric tenderness.

Laboratory investigations showed a white blood cell count of 19 (×109/l), serum albumin level of 2.4 g/dl, and slightly elevated tumor markers (carcinoembryonic antigen=7.6 ng/ml and CA19–9=65.5 U/ml). Abdomen ultrasonography revealed nonvisualized gallbladder due to gassy abdomen and dilated intestinal loops. Barium meal and follow through revealed abrupt stoppage of contrast at the duodenojejunal flexure with large filling defect and abnormal outpouch of the duodenum suggestive of cholecystoduodenal fistula, as shown in [Figure 1]. The provisional diagnosis was obstruction at the duodenojejunal flexure due to tumor or impacted foreign body, and hence the patient was scheduled for surgical exploration.
Figure 1 Oral contrast study showing abrupt stoppage of contrast at the duodenojejunal flexure with outpouch at the first part of the duodenum.

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Midline laparotomy was performed and exploration revealed dilated stomach, duodenum, and upper jejunum until large impacted stone at 10 cm from the duodenojejunal flexure and collapsed intestinal loops distally. The gallbladder was not seen due to marked adhesions between it and the omentum. Through a longitudinal enterotomy, a 3.5 cm stone was extracted from the first jejunal loop ([Figure 2]). Neither cholecystectomy nor repair of the cholecystoduodenal fistula was performed.
Figure 2 Intraoperative image showing a 3.5 cm gallbladder stone that was extracted from the first jejunal loop.

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The postoperative course was uneventful apart from wound infection treated conservatively with antibiotics. He was discharged on postoperative day 6 with good general condition.

Case 2

A 70-year-old diabetic woman presented to our outpatient clinic with 10-day history of persistent vomiting, upper abdominal pain, and absolute constipation. The patient gave a long past history of gall stone disease. Physical examination revealed no abnormalities apart from succussion splash over the upper abdomen.

Plain abdominal radiography revealed no evidence of small bowel obstruction or evidence of pneumobilia. Abdominal ultrasonography revealed no evidence of gall bladder stones. Abdominal computed tomography scan was performed and showed a large stone measuring 4 cm impacted in the third part of the duodenum with evidence of cholecystoduodenal fistula ([Figure 3]).
Figure 3 An abdominal computed tomography (CT) scan showing a large stone impacted in the third part of the duodenum and evidence of cholecystoduodenal fistula.

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Laparotomy was performed through a midline incision. After mobilization of the duodenojejunal flexure by dissection of Treitz ligament, milking of the stone to the first jejunal loop was carried out. Stone extraction through a longitudinal enterotomy was carried out ([Figure 4]). The gall bladder and the fistula were left alone.
Figure 4 Operative image of the stone after extraction.

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The patient passed a smooth postoperative course and was discharged on the third postoperative day.


  Discussion Top


First reported in 1896 by Leon Bouveret, Bouveret's syndrome is a rare form of gall stone ileus accounting for 1–4% of its cases. It is caused by migration of a large gall bladder stone through a cholecystoduodenal fistula and its impaction in the duodenum, causing an upper gastrointestinal obstruction [1],[2],[3].

Risk factors favoring the development of this syndrome include age more than 60 years, female sex, large-sized gall bladder stones (>2 cm), long history of gall stone disease, and repeated attacks of acute cholecystitis [3],[4].

Because of the nonspecific presentation of this condition in which more than 30% of the patients do not give a history of prior gall stone disease and are not of old age, diagnosis of Bouveret's syndrome may be a clinically challenging situation [5].

Plain abdominal radiography is of low value as the classical Rigler triad of small intestinal obstruction, pneumobilia, and ectopic calcified gall stone can be demonstrated in 10–50% of the cases. Abdominal ultrasonography may be helpful in demonstrating pneumobilia; however, it has its own limitations due to gastric and intestinal distension and anatomical alterations due to the presence of the bilioenteric fistula [2],[4].

Oral contrast study can be very helpful as demonstrated in our first case to detect the level and possible cause of obstruction. However, the best tool to diagnose Bouveret's syndrome is abdominal computed tomography scan. Although it can detect the presence of the stone in the duodenum with high accuracy, it can also demonstrate the anatomy of the bilioenteric fistula as shown in our second case [5].

Treatment goal of Bouveret's syndrome entails management of the intestinal obstruction through stone extraction. Various approaches are available for achieving this goal, including endoscopy, extracorporeal shock wave lithotripsy, and surgery.

Given the old age of the patients and coexisting serious morbidities, endoscopy provides a less invasive approach with a low rate of complications. Many endoscopic techniques have been reported, such as endoscopic removal, net extraction, mechanical lithotripsy, electrohydraulic lithotripsy, and intracorporeal laser lithotripsy [6]. Extracorporeal shock wave lithotripsy is reported to be one of the successful ways to manage Bouveret's syndrome, although multiple settings may be required and may be difficult to perform in obese patients or if there are gas-containing bowel loops between the gallstone and the abdominal wall [7].

Surgical enterotomy and stone extraction remain the classical therapeutic modality for Bouveret's syndrome, especially after failed endoscopic approach. Open surgery, laparoscopy, and endoscopically assisted minimally invasive surgery have been described [8]. In our report, the two cases underwent open surgery, as the position of the stone was out of reach for any endoscopic approach; moreover, advanced endoscopic facilities such as electrohydraulic lithotripsy were not available. Crushing of the stones in both cases was not possible due to their hard nature, which necessitated an enterotomy and stone extraction.

Concomitant cholecystectomy and fistula closure is a matter of controversy. Some authors advocate cholecystectomy and fistula closure to avoid persistent symptoms, recurrence, and possibility of gall bladder cancer. However, because of the higher morbidity and mortality rates in patients who underwent cholecystectomy and fistula closure (20–30%) compared with those with simple stone extraction (12%), most authors agree that stone extraction is enough, given the old age and coexisting morbidities of these patients [9]. In our two cases, no attempt was made to attack the gall bladder or the fistula.


  Conclusion Top


Because of the patient's old age, nonspecific presentation, and its rarity, Bouveret's syndrome is a clinically challenging diagnosis to make. Although surgical stone extraction remains the classic treatment option with the least failure rates, leaving the fistula alone is associated with the least morbidity and mortality rates.

Financial support and sponsorship

Nil.

Conflicts of interest

The authors declare no conflict of interest.

 
  References Top

1.
O’Neill C, Colquhoun P, Schlachta CM, Etemad-Rezai R, Jayaraman S. Gastric outlet obstruction secondary to biliary calculi: 2 cases of Bouveret syndrome. Can J Surg 2009; 52:E16–E18.  Back to cited text no. 1
    
2.
Iancu C, Bodea R, Al Hajjar N, Todea-Iancu D, Bălă O, Acalovschi I. Bouveret syndrome associated with acute gangrenous cholecystitis. J Gastrointestin Liver Dis 2008; 17:87–90.  Back to cited text no. 2
    
3.
Lawther RE, Diamond T. Bouveret's syndrome: gallstone ileus causing gastric outlet obstruction. Ulster Med J 2000; 69:69–70.  Back to cited text no. 3
    
4.
Masannat YA, Caplin S, Brown T. A rare complication of a common disease: Bouveret syndrome, a case report. World J Gastroenterol 2006; 12:2620–2621.  Back to cited text no. 4
    
5.
Brooks Brennan G, Rosenberg RD, Arora S. Bouveret syndrome. Radiographics 2004; 24:1171–1175.  Back to cited text no. 5
    
6.
Langhorst J, Schumacher B, Deselaers T, Neuhaus H. Successful endoscopic therapy of a gastric outlet obstruction due to a gallstone with intracorporeal laser lithotripsy: a case of Bouveret's syndrome. Gastrointest Endosc 2000; 51:209–213.  Back to cited text no. 6
    
7.
Dumonceau JM, Delhaye M, Devière J, Baize M, Cremer M. Endoscopic treatment of gastric outlet obstruction caused by a gallstone (Bouveret's syndrome) after extracorporeal shock-wave lithotripsy. Endoscopy 1997; 29:319–321.  Back to cited text no. 7
    
8.
Thompson RJ, Gidwani A, Caddy G, McKenna E, McCallion K. Endoscopically assisted minimally invasive surgery for gallstones. Ir J Med Sci 2009; 178:85–87.  Back to cited text no. 8
    
9.
Rodríguez-Sanjuán JC, Casado F, Fernández MJ, Morales DJ, Naranjo A. Cholecystectomy and fistula closure versus enterolithotomy alone in gallstone ileus. Br J Surg 1997; 84:634–637.  Back to cited text no. 9
    


    Figures

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



 

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