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CLINICAL CASES

Received: February 5^th 2015

Accepted: March 25^th 2015

Bouveret’s syndrome: A rare presentation of gallstone ileus

Luis Franco-Avilés,^a Héctor Tonalli Arce-Guridi,^a Ulises Mercado^a

^aHospital General de Mexicali, nstituto de Servicios de Salud Pública del Estado de Baja California

Mexicali, Baja California, México

Communication with: Ulises Mercado

Email: dr_omr_2012@yahoo.com

Background: Bouveret’s syndrome is defined as gastric outlet obstruction caused by duodenal impaction of a gallstone which passes into the duodenal bulb through a cholecystoduodenal fistula.

Clinical case: We reported the case of a 46-year-old woman who presented intermittent epigastric pain, nausea, vomiting, and weight loss of 2-months duration. The patient admitted alcohol and methamphetamine abuse. She had not fever, dehydration or jaundice. Amylase, electrolytes, enzymes, and creatinine level were within normal limits. Seven months previously she was seen in the emergency department for acute cholecystitis. In that occasion, an abdominal ultrasound was reported with cholelithiasis without dilatation of the intra/extra-hepatic bile duct. Abdominal plain radiographs showed no relevant findings. A laparoscopic surgery was performed. During the procedure a sub-hepatic plastron with firm adhesions was found. The gallbladder was found attached to the duodenal bulb and an impacted calculus in the duodenum. The procedure was converted to surgery. Surgeon decided to perform a Bilroth 1 as the best choice. Seven days later, she was discharged.

Conclusion: The clinical manifestations of the Bouveret’s syndrome are nonspecific. Preoperative diagnosis is a challenge for clinicians because of the rarity of this condition. Treatment must be individualized.

Keywords: Gallbladder; Gallstones; Bile duct diseases

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Bouveret syndrome (BS) is a rare variant of gallstone ileus, caused by the passage and impaction of a stone in the pylorus or duodenum, after passing through a cholecystoduodenal fistula, resulting in obstruction of the gastric outlet. Six risk factors are involved in the formation of BS: 1) history of cholelithiasis; 2) repeated episodes of cholecystitis; 3) female gender; 4) older than 60 years; 5) comorbidity, and 6) gallstones greater than 2.5 cm in diameter.¹

According to the medical literature, there were previous descriptions of what is now known as BS. In 1896, the French physician Leon Bouveret reported the cases of two patients with pyloric stenosis attached to the gallbladder. This syndrome occurs in 1-3% of cases of gallstone ileus.¹ We present the case of a young woman who came to the hospital with biliary colic, with no evidence of peritoneal irritation or gastric dilatation. Laparoscopic cholecystectomy was performed, but the presence of subhepatic plastron necessitated open exploration.

Clinical case

46-year-old woman who presented to the ER in September 2013 with intermittent abdominal pain 3 months in duration, as well as nausea, vomiting, and weight loss. She was an alcoholic and abused methamphetamines. The examination revealed diffuse abdominal pain without peritoneal irritation. Laboratory results, including enzymes, amylase, serum creatinine, and electrolytes, were normal. Abdominal ultrasound reported vesicular calculation without bile duct dilatation (Figure 1). Plain abdominal x-rays were not of diagnostic utility. She received intravenous fluids and antispasmodics and was discharged voluntarily. She went to the outpatient clinic in March 2014 with biliary colic with a history of eating cholecystokinin-triggering foods. She was scheduled for elective laparoscopic surgery. During surgery, multiple adhesions were found that complicated the procedure, so open surgery was elected. One plastron was found formed by a scleroatrophic gallbladder, omentum, and duodenum with firm adhesions. Upon recognizing the cyst, cholangiography was performed, finding a normal bile duct. There was mild gastric distension. An incision of 6 cm was made to the first portion of the duodenum and pylorus and 2 cm of the stomach, finding a stone of 2.5 cm, which was extracted (Figure 2). Due to the large inflammatory process, the risk of suture fistulation was evaluated, so it was decided to resection the antrum, pylorus, and 2-3 cm of the first portion of duodenum as a Bilroth 1 anastomosis. The patient was discharged in good condition.

Discussion

BS is rarely documented in the medical literature. In 2006, Cappell and Davis2 identified 128 cases of BS in world literature. The mean age was 74 ± 11 years, predominantly in women; nausea and vomiting occurred in 87%, abdominal pain in 71%, and weight loss in 14%. Just 4 isolated cases were reported in our country from 2004 to 2014.^3-6 The average age was 68.7 years, range 52 to 83. In three of the cases, the diagnosis was made by endoscopy and EGD series, however, they ended in open surgery. In the fourth case, laparoscopic surgery alone was successful.

Noriega-Maldonado et al.⁷ reviewed the cases of cholecystectomy and intestinal obstruction over a period of three years. The authors found 1054 cases of cholecystectomy, and 189 cases of intestinal obstruction in various age groups; of these, seven (3.7%) had intestinal obstruction. In the jejunum, two cases; in the ileum, four cases, and in the duodenum, one case. In the seven cases, diagnosis was made preoperatively (in five cases it was based on the findings of plain abdominal X-ray).

Our patient is a younger woman with a history of chronic cholecystitis, alcoholism, and drug use, without data of complete gastric outlet obstruction, gastric dilatation, or dehydration, and with clinical signs over several weeks. The plain abdominal X-rays were not helpful. The radiological report showed a stone in the gallbladder, when in fact it was in the duodenum; therefore, the preoperative diagnosis of BS could not be established. However, Rigler’s classic triad (pneumobilia, gallstone outside the gallbladder, and small bowel obstruction) is present in only 30 to 50% of cases.^8,9

There are several alternatives in the management of BS, but they are controversial. Most reports are isolated with a maximum of five cases,¹⁰ and conclusions cannot be drawn. Treatment must be individualized; in elderly the alternatives must be the least invasive.

 

References

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8. Mavroeidis VK, Matthioudakis DI, Economou NK, Karanikas ID. Bouveret syndrome-The rarest variant of gallstone ileus: A case report and literature review. Case Reports Surg 2013;2013:1-6.
9. Gajendran M, Muniraj T, Gelrud A. A challenging case of gastric outlet obstruction (Bouveret´s syndrome). J Med Case Reports 2011:5:1-4.
10. Rodríguez-Hermosa JI, Roig J, Girones J, Codina-Cazador A, Hombrados M. Síndrome de Bouveret: descripción de 5 casos. Cir Esp 2004;76:256-60.

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