Multidisciplinary management of Mirizzi syndrome with cholecystobiliary fistula: the value of minimally invasive endoscopic surgery
 
Fabien Le Roux, Charles Sabbagh, Brice Robert, Thierry Yzet, Laurent Dugue, Jean-Paul Joly and Jean-Marc Regimbeau
Amiens, France
 
Author Affiliations: Department of Digestive and Oncological Surgery (Le Roux F, Sabbagh C and Regimbeau JM); Department of Interventional and Diagnostic Imaging (Robert B and Yzet T); and Department of Hepatogastroenterology (Joly JP), Amiens University Hospital, Jules Verne University of Picardie, Amiens, France; Department of Digestive Surgery, Saint-Camille Hospital, Bry-sur-Marne, France (Dugue L)
Corresponding Author: Professor Jean-Marc Regimbeau, Department of Digestive and Oncological Surgery, Amiens University Hospital, Place Victor Pauchet, F-80054 Amiens cedex 1, France (Email: regimbeau.jean-marc@chu-amiens.fr)
 
© 2015, Hepatobiliary Pancreat Dis Int. All rights reserved.
doi: 10.1016/S1499-3872(15)60380-0
Published online May 21, 2015.
 
 
Contributors: RJM conceived the study. LRF and SC wrote the article. DL, RB, YT and JJP provided the iconography. RJM is the guarantor.
Funding: None.
Ethical approval: This study was approved by the local independent ethics committee CCPP Nord Est II, Amiens, France.
Competing interest: No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article.
 
 
ABSTRACT: Mirizzi syndrome, a rare complication of gallstones, is defined by obstruction of the main bile duct. This obstruction may worsen and thus result in cholecystobiliary fistula. Surgical management of Mirizzi syndrome is complicated by the presence of inflamed tissue around the hepatic pedicle, making it impossible to distinguish between the main bile duct and the gallbladder. The surgeon's first task is to perform subtotal cholecystotomy (from the fundus of the gallbladder to the neck) without trying to locate the cystic duct. In a second step, the gallstones are extracted and the main bile duct is then repaired. In most cases, a T-tube is used to drain the main bile duct, and abdominal drainage is left in place (in case a bile fistula forms). This study concluded that preoperative drainage of the main bile duct in the treatment of Mirizzi syndrome types II and III is feasible and might help to decrease the postoperative complication rate.
 
(Hepatobiliary Pancreat Dis Int 2015;14:543-547)
 
KEY WORDS: jaundice; Mirizzi syndrome; cholecystobiliary fistula; endoscopic treatment; cholecystectomy; T-tube drain
 
 
Introduction
Mirizzi syndrome is a rare complication of gallstones. It is a chronic complication that is defined by a main bile duct obstruction (caused by mechanical compression and inflammation of the surrounding tissue) after a gallstone becomes impacted at the neck of the gallbladder or in the cystic duct.[1, 2] In order to facilitate the choice of appropriate surgical management, many different classifications of Mirizzi syndrome have been developed. The Csendes classification is among the most frequently used (Fig. 1).[3-5] Csendes types II and III account for 40% of all cases of Mirizzi syndrome[6] and correspond to a cholecystobiliary fistula that involves less than one third of the circumference of the main bile duct (for type II) or up to two thirds (for type III).[6] The recommended treatment is main bile duct repair (without biliary digestive anastomosis) and, in most cases, external biliary drainage.[6]
 
External biliary drainage is associated with specific complications and a mean serious morbidity rate of 11.3%.[6-8] Thus, avoidance of external biliary drainage might decrease the overall morbidity rate.
 
The objective of the present single-center brief report was to establish whether a biliary drain (inserted via endoscopic retrograde cholangiopancreaticography (ERCP) was of value for (i) identifying the main bile duct and (ii) avoiding external biliary drainage in the management of Mirizzi syndrome type II or III.
 
 
Methods
All patients treated between November 2012 and January 2014 for Mirizzi syndrome type II or III (according to the Csendes classification) in the Department of Digestive and Oncological Surgery at Amiens University Hospital (Amiens, France) were retrospectively included. These inclusions were reviewed after approval by the local independent ethics committee (CPP Nord Est II, Amiens, France).
 
Diagnosis of Mirizzi syndrome
Mirizzi syndrome was suspected in patients presenting with right upper quadrant pain, cytolysis and cholestasis. A computed tomography (CT) scan was then performed. Mirizzi syndrome was diagnosed as obstruction of the main bile duct (caused by mechanical compression and inflammation of the surrounding tissue) after a gallstone had become impacted at the neck of the gallbladder or in the cystic duct.[9] On the basis of the CT data, Mirizzi syndrome was graded according to the Csendes classification.[3, 5] Magnetic resonance cholangiopancreaticography was not performed because (i) there were no suspected malignancies, (ii) a gallstone was evidenced by CT in all cases and (iii) Mirizzi syndrome was diagnosed adequately on the basis of the CT.
 
Surgery-only management of Mirizzi syndrome
A laparotomy procedure was performed. First, the gallbladder was opened and the gallstone was extracted. Anterograde subtotal cholecystectomy (without dissection of Calot's triangle) was then performed.[10, 11] Given that the main bile duct defect necessarily involves less than two thirds of the duct circumference in Mirizzi syndrome types II and III (according to the Csendes classification), the remnant gallbladder was used to repair the main bile duct. The abdominal cavity was drained by placing the drain close to the site of biliary duct repair.
 
Endoscopic and surgical management of Mirizzi syndrome
In two cases, cholangitis prompted us to perform ERCP (in order to reduce bilirubin levels, improve the patient's general condition and decrease the risk of sepsis during subsequent treatment).
 
Prior to surgery, an endoscopic stent or a nasobiliary drain was used to drain the main bile duct. Three days after insertion of the nasobiliary drain or six weeks after placement of the endoscopic stent, the bile duct was surgically repaired by using the remnant gallbladder. No external biliary drains (T-tubes) or abdominal drains were inserted. The internal drain was left in place and the bile duct was repaired, while leaving the drain or stent in place (thus avoiding the need for a T-tube). The main bile duct drain was then removed on postoperative day 5 (if a nasobiliary drain had been used after cholangiography) or at postoperative week 6 (in patients with a endoscopic stent).
 
 
Results
From November 2012 to January 2014, five patients with Mirizzi syndrome type II or III (according to the Csendes classification) were treated in our department. Three were treated with surgery only and two were treated with endoscopy and surgery.
 
All three "surgery-only" patients were discharged with a T-tube, which was removed at postoperative week 6. Before discharge, all patients underwent cholangiography on postoperative day 5 to check for the absence of bile leakage or bile duct dilatation. The early postoperative courses outcomes were uneventful in all cases. The mean length of stay was 8 days (range 7-9). After T-tube removal, two patients developed bilioperitonitis and required repeat surgery to wash and drain the abdominal cavity and ERCP with use of an endoscopic stent to close the main bile duct defect. During surgery, there was no fibrosis seen around the drain site and the no fibrous path could be the reason for peritonitis after T-tube removal. The biliary fistula was successfully repaired in patients 1 and 2 (Fig. 2).
 
The two "surgery plus endoscopy" patients had uneventful postoperative courses. No abdominal pain was noted and the liver function test results returned progressively to normal values. The mean length of stay was seven days. In one patient, the preoperative nasobiliary drain was removed on postoperative day 5, after cholangiography had been performed through the drain itself. The second patient was readmitted 6 weeks later for stent removal. The three-month follow-up period was also uneventful, with no bile leakage or inflammatory stenosis of the main bile duct (patients 4 and 5; Fig. 2). After discussion, the patient with the nasobiliary drain elected to keep it for five days (rather than undergo another operation under general anesthesia). The patient with the main bile duct drain had no preference when asked to choose between a nasobiliary drain and general anesthesia.
 
 
Discussion
Mirizzi syndrome is a rare complication of gallstones. Cholecystobiliary fistula increases the risk of main bile duct injury during cholecystectomy, and so preoperative screening for Mirizzi syndrome is an important step in decreasing this risk. It is noteworthy that although the syndrome is difficult to treat, it is easy to diagnose (based on right upper quadrant pain, cytolysis, cholestasis, an impacted gallstone in the Hartmann pouch or in the cystic duct, and main bile duct and intrahepatic duct dilatation on a CT scan).[9] Thus, a first-line CT scan can reveal gallstones and dilated bile ducts. Although MRI is more sensitive for the diagnosis of bile duct stones (88%-92%), biliary strictures (93%-100%) and biliary tract lesions (97%-99%), it is not always available in routine practice.[8]
 
As mentioned above, surgical management of Mirizzi syndrome is problematic. The frequent presence of dense adhesions and local inflammation means that dissection of the main bile duct is often difficult. These factors justify the insertion of a T-tube in almost all cases (and even in 100% of the cases described by Li et al).[1, 6] We have previously reported on the "endoscopy-only" management of Mirizzi syndrome in two patients.[12]
 
External biliary drains require specific care procedures; choledocotomy must be performed below the fistula, in order to introduce a T-tube for drainage of the main bile duct and thus avoid insertion of the drain into fragile tissue near the fistula.[5] Indeed, choledocotomy may be associated with complications, as observed in the present report. In a review, Lai and Lau[13] reported a postoperative morbidity rate of up to 60% in patients treated for Mirizzi syndrome. The rates were highest for types II, III and IV (relative to type I) but the impact of external biliary drainage was not specified. In the series reported by Erben et al,[1] the postoperative morbidity rate was 31%. Postoperative events included hemorrhage, residual gallstone, bile leakage, bile duct injury, external fistula, delayed-onset bile duct stricture and death; indeed, common bile duct leakage accounted for 9% of all complications. In a recent review, Ahmed et al[7] reported an incidence risk of bile peritonitis after T-tube removal of between 2.5% and 19.6% (with a risk of repeat surgery and death), although the reviewed series included some patients with conditions other than Mirizzi syndrome. Moreover, other studies[14-16] have reported that T-tubes increase the operating time and the length of stay.
 
The combination of endoscopic and surgical procedures facilitates preoperative identification of the main bile duct and postoperative management, and does not require specific care procedures. In some cases, "laparoscopy-only" procedures may be possible; Li et al[6] reported on endoscopic/laparoscopic procedures (insertion of a nasobiliary drain) in 27 patients with Mirizzi syndrome types I to III (according to the Csendes classification) and compared them with 27 patients treated with laparotomy and no preoperative ERCP. The researchers observed a shorter operating time, less blood loss, quicker initiation of oral refeeding and a shorter hospital stay in the endoscopic/laparoscopic group.
 
Use of a nasobiliary drain may be of value because it enables early postoperative cholangiography and drain removal. The length of stay is shorter and there is no need for upper digestive endoscopy under general anesthesia to remove the stent.
 
In conclusion, preoperative drainage of the main bile duct in the treatment of Mirizzi syndrome types II and III is feasible and might help to decrease the postoperative complication rate. This strategy might be especially indicated when the patient has cholangitis or poor general status. However, studies of larger series are needed before firm conclusions can be drawn.
 
 
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Received July 21, 2014
Accepted after revision January 9, 2015