Bouveret syndrome: simultaneous open surgery as definitive treatment in a young patient, a case report

Abstract

Introduction

Bouveret syndrome is an uncommon complication of cholelithiasis in which a large gallstone causes an impacted intestinal obstruction. This stone migrates through a cholecystenteric fistula formed by chronic inflammation and adhesions, which increase intraluminal pressure, leading to ischemia and subsequent perforation [1].

Historically, it is a rare medical condition with only a few hundred cases reported globally from the last century to the present day. Although its diagnosis has become more accessible due to advanced imaging techniques available in most secondary and tertiary care hospitals, it still represents a diagnostic challenge and accounts for <3% of all gallstone ileus cases.

Its clinical presentation is nonspecific and easily confused with more common pathologies; it does not generate specific biochemical alterations to guide the diagnosis, and Rigler's classic triad (pneumobilia, ectopic gallstones, and intestinal obstruction) is observed in only approximately half of the patients. Due to these circumstances, diagnosis can be significantly delayed, increasing the mortality risk up to 30% due to late surgical intervention [2–4].

This report presents the case of a female patient with symptoms suggestive of cholecystitis who was diagnosed with Bouveret syndrome after diagnostic investigations.

Case report

A 48-year-old female was admitted to the emergency department presenting with abdominal pain in the right hypochondrium, nausea, vomiting, and diarrheal stools following food intake, with a 3-day onset. Her only significant medical history was a bilateral tubal occlusion 20 years ago. Vital signs were: heart rate: 90 beats per minute; blood pressure: 120/70 mmHg; respiratory rate: 18 breaths per minute; temperature: 37°C; oxygen saturation: 93%.

The general surgery service requested complementary examinations for preoperative evaluation under the suspected diagnosis of acute-on-chronic cholecystitis. An ultrasound revealed a gallbladder containing a gallstone, and a chest X-ray showed pneumobilia. Given these findings, an abdominal computed tomography (CT) scan was requested (Fig. 1), demonstrating two ectopic gallstones in the duodenum. The diagnosis of Bouveret syndrome with a cholecystenteric fistula and secondary intestinal occlusion was established.

Figure 1

Abdominal CT, an ovoid structure measuring 49 mm compatible with an ectopic gallstone is identified, along with air bubbles in the biliary tract consistent with pneumobilia.

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An exploratory laparotomy was performed for gallstone extraction (Fig. 2), along with a cholecystectomy, pyloric exclusion, a side-to-side jejuno-jejunostomy, and a Roux-in-Y gastrojejunostomy (Fig. 3).

Figure 2

Extraction of the gallstone.

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Figure 3

Side-to-side jejuno-jejunostomy with pyloric exclusion and Roux-in-Y reconstruction.

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After 10 days of postoperative care, the patient was discharged with a favourable evolution.

Discussion

Bouveret syndrome is a highly infrequent entity caused by intestinal obstruction due to a gallstone that migrates through a cholecystenteric fistula. In this clinical case, it occurred in a middle-aged woman, contrasting with most reported cases where the mean age at diagnosis is between 74 and 78 years. With a single history of bilateral tubal occlusion—considered a minor surgery—the factors most associated with this condition are major surgeries, such as partial gastrectomies or Billroth II reconstructions for gastric cancer or duodenal ulcers, making this patient's presentation unusual [1, 5–8].

This entity represents a diagnostic and therapeutic challenge for the surgeon, as there is no consensus or standardized guideline regarding the choice of initial treatment, the extent of the surgical intervention, and whether to perform a single-stage or two-stage surgical repair.

Part of the literature points to endoscopy as a first-line intervention because it is a minimally invasive procedure; however, its success rate is 43% compared to open surgery, which was chosen for this patient, yielding success rates exceeding 90% [1, 9].

An advanced surgical approach was selected due to the damage caused by both stones to the duodenal wall; consequently, pyloric exclusion and a Roux-en-Y gastrojejunostomy were performed to prevent leaks due to tissue friability or stenosis. The clinical characteristics of this patient allowed for a favourable outcome by utilizing open surgery as the first option and performing a single-stage surgical procedure, extracting the stones and repairing the fistula simultaneously—a technique used to prevent recurrences, cholecystitis, and the potential risk of developing cancer.

Each case must be individualized. In elderly patients, endoscopy could be considered a first-line treatment despite its low success rate due to the high risk of complications, performing the fistula closure and cholecystectomy in a second surgical stage. In patients with comorbidities representing a very high risk, conservative management can be chosen, as literature describes spontaneous closure of the fistula [9].

Bouveret syndrome is a very rare complication of cholelithiasis. In this case, resolution was achieved through single-stage open surgery, which allowed for complete remission. However, in high-risk patients with comorbidities, endoscopy might be preferred as the sole measure or a reoperation for fistula repair if the patient's conditions permit. Each case must be individualized based on patient characteristics and available resources.

The documentation of such an infrequent entity and its method of resolution is essential as a precedent for future references.

Conflicts of interest

None declared.

Funding

None declared.

References