J Gastrointestin Liver DisMarch 2008 Vol. 17 No 1, 87-90Address for correspondence: Assoc. Prof. Dr. Cornel Iancu Clinica Chirurgie III Str. Croitorilor, Nr. 19-21 400162, Cluj-Napoca, Romania Email: dr_cornel_iancu@yahoo.com

Bouveret Syndrome Associated with Acute Gangrenous CholecystitisCornel Iancu, Raluca Bodea, Nadim Al Hajjar, Dana Todea-Iancu, Ovidiu Bălă, Iurie Acalovschi

3rd Surgical Clinic Cluj-Napoca, University of Medicine and Pharmacy, Cluj-Napoca, Romania

AbstractAn 89-year-old patient was hospitalized with signs of

acute lithiasic cholecystitis and gastric emptying failure. The decision for surgery was taken and a subhepatic block was evidenced, caused by a perforated gangrenous cholecystitis with pericholecystic abscess, a cholecysto-antroduodenal fistula with two gallstones, 9/5 and 4/3 cm in size, impacted in the duodenum. It was necessary to perform an Y-en-Roux antroduodenojejunal anastomosis because an antroduodenal parietal defect resulted after the removal of the gangrenous gallbladder. The immediate and long term postoperative evolution in terms of anastomosis functionality was good.

Key wordsGangrenous cholecystitis – Bouveret syndrome – Y-en-

Roux antroduodenojejunal anastomosis

IntroductionThe Bouveret syndrome, described for the first time

in 1896 by Leon Bouveret, is defined as a high intestinal obstruction caused by the impaction of one or more gallstones in the duodenum, consecutive to a biliodigestive fistula [1, 2]. It is a particular form of biliary ileus, occurring in 3% of all cases of gallstone impaction in the digestive tract, which represent 1-3% of all intestinal obstruction causes [3].

This fact is also evidenced by the relatively few cases reported in the literature, up to 300 published cases [4, 5].

In a patient suspected of Bouveret syndrome, it is essential to establish a positive diagnosis and to perform an emergency therapeutic intervention for the subsequent good evolution.

These are the most important factors which contribute to the decrease of mortality, estimated at 15-25% [1].

Case ReportAn 89-year-old female patient, with no significant

personal history, was admitted to the 3rd Surgical Clinic Cluj, complaining of pain in the right hypochondrium, fever, nausea, loss of appetite and abdominal distension.

The patient was in a good condition, but with pale skin and mucosae, had hemodynamic and respiratory stability, diuresis and intestinal transit were present. The abdominal examination evidenced an inflammatory subhepatic block and sensitivity to right upper quadrant palpation.

Laboratory examinations, except a slight increase in amylasemia (315 U/l)(normal values up to 200 U/l) and leucocytosis (11,700 K/uL), were within normal limits.

Abdominal ultrasound examination revealed the liver at the upper limit of normal, an enlarged gallbladder, filled with gallstones, and a 5 mm transonic area around the gallbladder. The ascending colon was adherent to the liver, aerocolia and aeroenteria were present. The surgical intervention

Fig.1 Macroscopic appearance of the gallstone causing the bilio-antroduodenal fistula.

88 Iancu et al

involved by the fistula and the pre-existence of stenotic areas at this level. Thus, the gallstone can be asymptomatic and can be eliminated with faeces or by vomiting, or it can become impacted somewhere along its digestive tract course, resulting in obstruction (15% of cases) [8]. In decreasing order of frequency, the obstruction can be located in the terminal ileum (50-75% of cases), the proximal ileum and the jejunum (20-40%), the colon, and more rarely, the stomach or the duodenum [9].

The therapeutic strategy is chosen taking into consideration several aspects: the patient’s age, comorbidities, the effect of obstruction on the general condition, the size of the gallstone and fistula, local inflammatory changes, the location of the obstruction. It has been demonstrated that the duration of the surgical intervention does not influence subsequent evolution, but evolution can be influenced by the delay in establishing the correct diagnosis and the unjustified delay in intervention [10].

The ideal treatment is to solve the obstruction by removing the gallstone, to close the fistula and prevent recurrences, and this can be achieved by open surgery.

If the extraction of gallstones whether by classic or laparoscopic duodenotomy or by an endoscopic procedure is compulsory at first, cholecystectomy and the solution of the fistula at another time or in serial interventions [5] are decided by the operative team. The approach will be mainly oriented by the general state of the patient and by the skills of the surgical team.

The preference of some surgeons for the intervention limited to duodenotomy with the extraction of gallstones should not be generalized. This approach will only be adopted for patients in an extremely poor state or in cases in which local inflammatory changes make the intervention extremely

was decided.Intraoperatively, a subhepatic block was evidenced. Its

dissection showed a perforated gangrenous gallbladder, with a pericholecystic abscess, a cholecysto-antroduodenal fistula, with two gallstones, 9/5 and 4/3 cm in size (Fig. 1), impacted at the antroduodenal level. After removal of the gangrenous gallbladder, a parietal defect with a length of about 11 cm was found at the level of the antrum and duodenum. The preferred solution was a side-to-side Roux-en-Y antroduodenojejunal anastomosis (Figs. 2, 3).

The patient was discharged on postoperative day 8. The immediate postoperative evolution, as well as the long term evolution were favorable. Barium examination at 6 months showed a functional anastomosis (Fig. 4).

DiscussionOne of the rarest complications of biliary lithiasis is

biliodigestive fistula, with a frequency of less than 1% (6). The factors favoring the fistula formation are: size of the gallstone (2-8 cm) [2], long history of biliary disease, repeated episodes of acute cholecystitis, female sex and advanced age (over 60 years) [1]. The fistula can be cholecystoduodenal (in 60% of cases), cholecystocolic (in 17%), cholecystogastric (in 5%), and choledocoduodenal (in 5%) [7].

Once in the intestinal lumen, the gallstone will manifest differently, depending on its size, the digestive tract segment

Fig.2 Posterior cross-section of the Roux-en-Y antroduodenal jejunal anastomosis.

Fig.3 Closure of the antroduodenal parietal defect by a L-L Roux-en-Y antroduodenal jejunal anastomosis; final appearance of anastomosis.

Fig.4 Radiological follow-up at 6 months; good passage of barium substance through the antroduodenal jejunal anastomosis.

Bouveret syndrome 89

difficult, predisposing to intraoperative complications. The supporters of simple interventions justify their decision by an increase in mortality when cholecystectomy and closure of fistula are associated, in 20-30% [11] compared to 12% in simple duodenotomy cases [9] and also by the fact that a cholecystoduodenal fistula may function as a biliodigestive anastomosis in the presence of a permeable cyst. The persistence of the fistula orifice favors stasis at the level of the terminal common bile duct and, secondary to it, cholangitis and lithiasis of the main bile duct [12, 13]. The closure of the fistula orifice leaves a dysfunctional scleroatrophic gallbladder, which favors the recurrence of lithiasis, acute cholecystitis or gallbladder cancer [14, 15]. In 1961, Bossart et al reported a 15% incidence of gallbladder carcinoma in patients with biliary fistula, as compared with only 0.8% in cholecystectomy samples at that time [16]. This group of patients at high risk also includes patients with Bouveret syndrome, in whom the fistula has not been closed. The postoperative persistence of symptoms in the majority of patients with duodenotomy and extraction of gallstones is another argument for performing cholecystectomy [15] in patients with a good general state.

The first intervention with the concomitant achievement of the three treatment objectives was performed in 1929, when Holz, trying to mobilize a gallstone impacted in the duodenum, involuntarily penetrated through the fistula orifice in the gallbladder and, after removing the gallstone, had to close the fistula tract and perform a cholecystectomy [17].

Sometimes, the solution of the parietal defect at duodenal level consists of a simple rupture, but it can also be a challenge to the surgeon, who may be obliged by the local situation to use other methods. The morbidity and mortality rates are determined to a large extent by the quality of the solution of the duodenal parietal defect.

Most authors report the suture of the fistula orifice [13]. In 1972, Redding et al performed vagotomy and Jaboulay gastroduodenostomy forced by the intense local inflammation, in a case of high intestinal obstruction due to impaction of two gallstones, one of 25 mm in the antrum and the other of 20 mm in the duodenal bulb [12].

With the accumulation of experience and the acquisition of adequate equipment, the laparoscopic approach, although difficult, has become possible. Sica et al [5] report the first case of Bouveret syndrome solved by laparoscopic route.

In well selected patients with important comorbidities and major surgical risk, less aggressive methods can be attempted, such as endoscopic procedures, which however will not correct the fistula and will not prevent recurrences. At endoscopy, the gallstone can be removed mechanically, electrohydraulically or by laser lithotripsy. Dumonceau et al have reported two cases treated with combined extracorporeal shock wave lithotripsy (ESWL) and electrohydraulic lithotripsy [18]. Maiss et al published two Bouveret syndrome patients successfully treated by laser lithotripsy, of whom the last one with a small sized fistula identified at bulbar level was followed up until its closure [19, 20].

In our patient, the association of Bouveret syndrome with a gangrenous fistula prompted us to perform cholecystectomy. The antroduodenal parietal defect, 11 cm in length, with the absence of the anterior duodenal wall, made the simple suture impossible, and therefore we chose a side-to-side Y-en-Roux antroduodenojejunal anastomosis. The control by barium examination at 6 months postoperatively showed good passage through the Roux loop, without barium opacification of the proximal duodenum and jejunum. This type of anastomosis was performed by our team in another case of Bouveret syndrome and in five cases of colon cancer with duodenal invasion, in all cases the anastomosis being well tolerated.

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