Endoscopic and Surgical Management of Bouveret Syndrome: Gastric Outlet Obstruction From an Impacted Gallstone

ABSTRACT

Impacted gallstones in the stomach and the duodenum lead to a rare presentation of gastric outlet obstruction known as Bouveret syndrome. Diagnosis and management is often challenging because of lack of streamlined protocol. However, when a diagnosis is made, there is an extensive toolkit available to endoscopists and surgeons to ensure favorable outcomes for the patient. In this article, we present a challenging case of Bouveret syndrome that required multidepartmental coordination and intervention.

INTRODUCTION

Bouveret syndrome is a rare cause of gastric outlet obstruction caused by a lodged gallstone in the stomach or duodenum. It was first reported in the 1700s and contributes to 2%–3% of gallstone-related gastrointestinal tract obstructions.¹ The stone reaches the small bowel through a bilioenteric fistula that forms as a result of chronic inflammation. Symptoms are generally nonspecific, and the diagnosis usually comprises of a combination of clinical presentation, radiological modalities, and endoscopy.² This condition is associated with high morbidity and mortality, especially as it predominantly affects elderly women and is often diagnosed late, highlighting the importance of early diagnosis and management.³ Only about 315 cases have been reported in the literature, and there are no standard guidelines for diagnosis or management.^4,5 We report a case of Bouveret syndrome managed with electrohydrolithotripsy, endoscopic ultrasound–guided gastrojejunostomy tube, and ultimately Roux-en-Y gastrojejunostomy.

CASE REPORT

A 55-year-old woman with a history of insulin-dependent diabetes presented with severe abdominal pain, associated with nausea and vomiting. Laboratory testing was notable for mild leukocytosis (11,000/μL), stable anemia (10.2 g/dL), elevated liver biochemistry profile (aspartate aminotransferase 60, alanine aminotransferase 62, and alkaline phosphatase 571 IU/L) and hyperbilirubinemia (5.6 mg/dL) with direct bilirubinemia (3.8 mg/dL). Right upper-quadrant abdominal ultrasound demonstrated air within the gallbladder fossa and within the wall of the gallbladder, and an abdominal computed tomography scan with oral and intravenous contrast revealed a 4-cm calcified gallstone eroding into the wall of the gallbladder adjacent to the antrum and duodenal bulb along with pneumobilia along with features of gangrenous cholecystitis (Figures 1 and 2). The patient then developed signs of sepsis with Gram-negative bacteremia, and therefore, broad-spectrum antibiotics were initiated. An esophagogastroduodenoscopy was performed, revealing a large gallstone eroding through a cholecystoduodenal fistula and completely obstructing the duodenal bulb. Hence, endoscopic retrograde cholangiopancreatography could not be performed (Figure 3). In an attempt to facilitate gallstone dislodgement and to provide temporary internal gallbladder drainage, electrohydrolithotripsy was performed. Despite using multiple probes, only a small fragment of the stone was dislodged, allowing guidewire-assisted placement of 2 double pigtail plastic stents extending from the stomach to the gallbladder. Purulent drainage immediately ensued. A nasogastric tube was placed under endoscopic guidance to facilitate gastric decompression. After discussion with the patient and the surgical team, it was decide to pursue endoscopic ultrasound (EUS)–guided gastrojejunostomy tube to facilitate gallbladder drainage and to establish a nutrition route bypassing the obstruction. The location of the stone complicated the surgical planning; therefore, this temporizing measure was pursued in the hope for the stone to passively dislocate.

Figure 1.

Right upper-quadrant ultrasound showing pneumobilia (blue arrow) and thickened gallbladder wall.

Figure 2.

Gas in the gallbladder concerning for emphysematous/gangrenous cholecystitis (blue arrow), and 4-cm gallstone eroded from the gallbladder wall into the duodenum (red arrow).

Figure 3.

Five to 6-cm stone in the duodenal bulb (A and B). Electrohydrolithotripsy (C). Two pigtail stents 7 × 12 Fr for internal drainage (D).

First, a 0.035-inch Jagwire was advanced to the side of the large gallstone into small bowel under endoscopic and fluoroscopic guidance. The wire was allowed to coil in the jejunum. Then a 9- to 12-mm balloon catheter was advanced into the jejunum, and the placement was confirmed by contrast injection. Next a 8.5 French nasocystic tube was advanced over the guidewire, and the guidewire was removed. Then, a solution of diluted contrast with saline and methylene blue was irrigated extensively into the jejunum, and EUS examined the stomach to identify a loop of jejunum. Once selecting an appropriate position in the stomach, the common wall between the stomach and jejunum was interrogated using a color Doppler. The stomach and jejunal wall were punctured under EUS guidance using electrocautery device while applying current to create G-J. A 20- × 10-mm AXIOS stent was placed (Figure 4).

Figure 4.

(A) 0.035-inch Jagwire was advanced to the side of the large gallstone in the duodenal bulb into the small bowel. (B) Contrast was injected confirming placement in the jejunum. (C) LAMS placed using electrocautery and current. (D) Gastric view of the LAMS. LAMS, lumen-apposing metal stent.

The patient then developed melena with a 3-g hemoglobin drop 2 days after the procedure. Emergent angiography revealed migration of the stone to the ligament of Treitz and bleeding in the gallbladder lumen in the venous phase (Figure 5). An upper endoscopy revealed dislodgement of the large gallstone and oozing from a large ulcer at the site of the cholecystoduodenal fistula (Figure 6). This was not amenable to endoscopic intervention; therefore, the surgical team was notified.

Figure 5.

Bleeding in the gallbladder in the venous phase (orange arrow). Interval migration of the stone to the ligament of Treitz (red arrow).

Figure 6.

Esophagogastroduodenoscopy showing bilioenteric fistula and ulceration around the site.

The surgery, in our case, was a 2-part process. During the first exploratory laparotomy, the stone was impacted in the duodenum, which was removed through a horizontal duodenotomy and closed primarily. The procedure was not completed because of intraoperative bleeding and subsequent hemorrhagic shock. The next day, during the secondary exploratory laparotomy, distal to the initial duodenal repair (in the D3-D4 region), there was another small perforation in the duodenum with surrounding necrosis, which was likely secondary to pressure injury from the stone. The duodenum appeared friable; therefore, the primary repair of this perforation was of questionable integrity. Decision was made to leave this small perforation and perform a Roux-en-Y gastrojejunostomy and a pyloric exclusion. She recovered well and was discharged to rehabilitation in good condition.

DISCUSSION

Bouveret syndrome is often missed in clinical practice because of its rare frequency and vague clinical presentation. There is no standard algorithm for management of Bouveret syndrome this rare entity. Our case emphasizes the necessity of early diagnosis and immediate intervention when there is a high clinical suspicion. This case also highlights the role of endoscopy in the management of this syndrome, particularly establishing endoscopic gallbladder drainage, and gastric obstruction bypass through an EUS-guided gastrojejunostomy using a lumen-apposing metal stent. It is important to highlight the role of a multidisciplinary discussion, which includes the patient, when making management decisions. Our patient wished to initially avoid surgery. Given her clinical presentation with sepsis and bacteremia, it was felt that drainage and antibiotics would optimize her condition before potential surgical intervention. As such, endoscopic interventions, including manipulating the gallstone back into the gallbladder, mechanical or electrohydraulic lithotripsy, and intracorporeal laser lithotripsy of the large stone, can be attempted first as they are less invasive and are associated with fewer complications in this largely elderly population.^6,7 However, up to 40% of patients fail endoscopic management. In addition, if the resultant fistula is large enough, it may not heal without surgical intervention. It also does not allow for the repair of the fistula.⁸ If endoscopic measures fail, or if the gallstone migrates after dislodgement, surgical interventions to remove the stone and repair the fistula are attempted. In our case, repeat endoscopic retrograde cholangiopancreatography was considered; however, given the clinical deterioration, the need for nutrition, and the development of gastrointestinal bleeding, surgical intervention was eventually pursued. Surgery can include enterolithotomy or gastrostomy, sometimes with cholecystectomy and/or fistula repair.⁹ Roux-en-Y surgery has been attempted in rare circumstances, such as in our case.¹⁰

Although Bouveret syndrome is an uncommon entity, it should be considered in patients with a history of gallstones and concerns for gastric outlet obstruction and be acted on promptly. In cases of larger stones, endoscopic diagnosis followed by multidepartmental discussion between gastroenterology and surgery teams should be pursued early to ensure the best outcomes for the patient.

DISCLOSURES

Author contributions: A. Manivannan and MZ Khan wrote the manuscript and reviewed the literature; S. Simmer, A. Watson, T. Zuchelli, and M. Elatrache reviewed and revised the manuscript for intellectual content; A. Manivannan, MZ Khan, and M. Elatrache revised the images; MZ Khan is the article guarantor.

Financial disclosure: T. Zuchelli is a consultant for Boston Scientific.

Informed consent was obtained for this case report.