A Multidisciplinary Approach to Management of Bouveret Syndrome

Abstract

Bouveret Syndrome is a rare complication of gallstone disease that occurs when a gallstone enters the stomach or bowel through a biliary enteric fistula and becomes impacted, resulting in gastric outlet obstruction. It is frequently seen in elderly chronically ill patients with neglected biliary disease. We describe a multidisciplinary approach to management of Bouveret Syndrome that could be adopted by healthcare systems with resources commonly found in facilities with a general urologist and gastroenterologist or general surgeon. Successful application of laser lithotripsy under endoscopic guidance sufficiently fractured the stone to allow for disimpaction and relief of the gastric outlet obstruction.

Bouveret Syndrome, first described in 1896 by Leon Bouveret,¹ is a rare variant of gallstone ileus. The condition occurs when a large gallstone enters the stomach or bowel through a biliary enteric fistula and lodges in the proximal duodenum, resulting in gastric outlet obstruction. The stone can be seen on esophago-gastroduodenoscopy in only approximately 69% of cases, owing to frequent submucosal embedding of the stone.² Endoscopic retrieval is rarely successful, although it should be attempted for patients with serious comorbidities.

Open or laparoscopic surgery is usually necessary and is often accomplished by enterotomy with stone retrieval and primary repair of the enterotomy.² Nonsurgical approaches to the treatment of Bouveret Syndrome have been described in the literature,^3–6 although Bouveret Syndrome is a rare entity so no large series have been reported. Here, we report successful treatment of Bouveret Syndrome using laser lithotripsy under endoscopic visual guidance.

Case Report

A woman, aged 45 years, presented with abdominal pain, nausea, and vomiting. She had a number of comorbidities, including cirrhosis (Child class B) with ascites resulting from alcohol abuse, esophageal varices from portal hypertension, poorly controlled diabetes, Lynch Syndrome, and active tobacco use. A year earlier, the patient had been diagnosed with cholecystitis, but owing to active alcohol use she was believed to be a poor surgical candidate and was instead treated with antibiotics. The patient reported that she had 5 to 6 episodes of cola-colored emesis daily and complained of increased lightheadedness. She also had constant right upper quadrant abdominal pain, the severity of which waxed and waned. The pain was not associated with meals.

Upon physical examination, the patient’s body temperature was 36.7°C (98°F), blood pressure was 99/71 mm Hg, heart rate was 117 beats per minute, and respiratory rate was 16 breaths per minute. She was alert, cooperative, and in no distress. She had no scleral icterus or jaundice. She was tachycardic but appeared to be well perfused. Her abdomen was scaphoid, soft, with right upper quadrant tenderness but without Murphy sign.

Results of the patient’s laboratory tests were as follows: white blood cell count 13,300/μL; glucose 551 mg/dL; sodium 118 mEq/L; chloride 73 mEq/L; potassium 3.9 mEq/L; creatinine 1.15 mg/dL which was increased from a baseline of 0.52 mg/dL; international normalized ratio (INR) of 1.3; albumin 2.9 g/dL; and alkaline phosphatase 3.84 U/L. Système International (SI) conversion factors for all relevant analytes to which this article refers are provided after the Conclusion. A right upper quadrant ultrasonogram showed numerous gallstones, a thin-walled gallbladder, negative sonographic Murphy sign, and a large stone in the proximal pylorus/stomach (Figure 1).

Figure 1.

Ultrasonogram revealing presence of large stone in the duodenum.

The patient was started on intravenous fluids and antibiotics, was kept non per os and was admitted to the hospital. A general surgeon was consulted and concluded that the patient was a high-risk surgical candidate due to her underlying Childs Pugh Class B cirrhosis and calculated postoperative mortality of 30%. After further consultation with gastroenterology, a minimally invasive approach to removal of the obstructing stone was chosen. The patient was taken to the lithotripsy suite, where shockwave lithotripsy was used in an unsuccessful attempt to break the stone. Next, a standard urologic laser lithotripsy setup was used under endoscopic visual guidance (Figure 2). The laser lithotripsy was performed with a 100-watt holmium laser (VersaPulse PowerSuite, Lumenis, San Jose, CA) with a 1000-micron fiber laser. Power began at the lowest setting and rapidly increased to 0.9 joules at a rate of 5 Hz per second. The stone then fractured into sufficiently small pieces to allow endoscopic removal with wire basket.

Figure 2.

Endoscopic view of the obstructing stone.

Discussion

Fifty percent of patients with gallstone ileus will report having had prior gallstone attacks, and 20% to 30% will have concomitant cholecystitis.⁷ Eighty-five percent of patients are women with a median age of 70 years.⁷ Owing to the comorbidities common to this patient population and to the severity of disease, gallstone ileus carries a modern mortality rate of 15% to 18%.⁷ Treatment typically consists of surgical lithotomy with delayed cholecystectomy, although a single-stage procedure is sometimes used in a subset of healthier patients because the spontaneous closure rate of biliary enteric fistula is relatively high. The exception to this paradigm may be cholecystocoloic fistula with the high concentration of bacterial reflux into the biliary system, resulting in severe cholangitis.⁷

Nonsurgical approaches to the treatment of Bouveret Syndrome have been reported. Gemmel et al³ described the use of a combination of mechanical lithotripsy, electro-hydraulic lithotripsy, and intracorporeal hydraulic laser lithotripsy. Staiano et al⁴ reported some success using a hydrojet lithotripsy system, and Langhorst⁶ was able to use a fluoroscopically-guided rhodamine laser for successful lithotripsy. In 2002, Alsolaiman⁵ reported using a holmium laser over the course of several separate procedures, which eventually broke down the stone for extraction using an endoscope.

Conclusion

We describe a multidisciplinary team approach to the care of a patient whose comorbidities rendered her a poor surgical candidate. Care provided involved team members and equipment that would be found at most hospitals with surgical capabilities. Bouveret Syndrome is a rare clinical entity that often occurs in medically ill individuals who are poor operative candidates. In the search for nonoperative options, numerous therapies, including urologic lithotripsy and biliary hydro lithotripsy, have been adopted from other fields. We successfully used laser lithotripsy under endoscopic guidance. The technology employed is common and could be found in hospitals with both a general urologist and general surgeon or gastroenterologist and offers a minimally invasive option to attempt in poor operative candidates who would otherwise face a potentially morbid operative intervention.

Système International Conversion Factors

Albumin: to convert to g/L, multiply value by 10.

Alkaline phosphatase: to convert to μkat/L, multiply value by 0.0167.

Chloride: to convert to mmol/L, multiply value by 1.0.

Creatinine: to convert to μmol/L, multiply value by 88.4. Glucose: to convert to mmol/L, multiply value by 0.0555. Potassium: to convert to mmol/L, multiply value by 1.0. Sodium: to convert to mmol/L, multiply value by 1.0. White blood cell count: to convert to ×10⁹/L, multiply value by 0.001.