Successful Treatment of Nonhealing Biliary-Cutaneous Fistulas With Biliary Stents

Transcription

1 GASTROENTEROLOGY 1986;90:764-9 Successful Treatment of Nonhealing Biliary-Cutaneous Fistulas With Biliary Stents ALAN C. SMITH, ROBERT H. SCHAPIRO, PETER B. KELSEY, and ANDREW 1. WARSHAW Departments of Medicine (Gastrointestinal Unit) and Surgery, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts Biliary-cutaneous fistulas occasionally complicate biliary tract surgery. Distal biliary obstruction, extensive loss of bile duct wall, or infection may contribute to the failure of such fistulas to heal. Five cases are reported of high-volume persistent fistulas that healed promptly after placement of endoscopic (4 cases) or percutaneous (1 case) biliary stents. Biliary stent placement eliminated the need for difficult reoperations in these complex patients and offers a promising therapeutic approach to this problem. Biliary-cutaneous fistulas are unusual complications of biliary tract disease. Though spontaneous biliarycutaneous fistulas have been described in the setting of long-standing untreated biliary tract disease, more commonly, such fistulas occur as complications of cholecystectomy or common duct exploration, as a result of surgical trauma to the extrahepatic bile duct, or as a consequence of local infection. Distal obstruction of the common bile duct is often present. We report here 5 cases of persistent high-volume postoperative biliary-cutaneous fistulas treated with endoscopically placed biliary endoprosthesis (4 of 5 cases) or percutaneous biliary stent (1 of 5 cases). In each case, fistulous drainage ceased shortly after stent placement and did not recur when the stents were eventually removed. Received July 18, Accepted September 17, Address requests for reprints to: Andrew L. Warshaw, M.D.. Massachusetts General Hospital, 15 Parkman Street, ACC 336, Boston, Massachusetts This work was supported by United States Public Health Service Training grant AM The authors thank Kristin Ahlman for her expert help in the preparation of this manuscript by the American Gastroenterological Association /86/$3.50 Materials and Methods From August 1984 to March 1985, 5 consecutive patients with nonhealing biliary-cutaneous fistulas were evaluated at the Massachusetts General Hospital (MGH). These patients represent the authors' entire experience during this period. The features of the 5 cases are summarized briefly in Table 1. The endoscopic procedures were performed with Olympus JFl T and J3.7 side-viewing endoscopes (Olympus Corporation of America, New Hyde Park, N.Y.), using light sedation with intravenous diazepam and meperidine, and all procedures were done with fluoroscopic monitoring. Endoscopic stents and introducer kits were from Wilson-Cook Company (Winston Salem, N.C.). Endoscopic papillotomies were carried out with the Cameron-Miller papillotome (Chicago, Ill.) using blended electrical current (electrocautery power source, Valley Lab, Boulder, Colo.). The percutaneous biliary drain placed in case 5 was done using the one-stick introducer system of Medi-Tech Inc. (Watertown, Mass.), and the 8.3F polyethylene catheter was made by Cook Inc. (Bloomington, Ind.). Case 1 A 47-yr-old man with steroid-dependent asthma was referred to the MGH with a biliary-cutaneous fistula persisting 1 mo after cholecystectomy for acute cholecystitis and cholelithiasis. During the initial surgery, an inadvertent Z-cm hole was made in the common hepatic duct. The nondilated bile duct was closed over a #10 T tube. One week later, a cholangiogram showed a patent bile duct without stones, but there was some extravasation of contrast around the T-tube entry site. The T tube was clamped on day 10, but 2 days later diffuse bile peritonitis led to reexploration. Breakdown of the previous bile duct repair was identified and treated with a trans duodenal transampullary choledochal stent and a subhepatic sump Abbreviation used in this paper: MGH, Massachusetts General Hospital.

2 March 1986 NON HEALING BCF WITH BILIARY STENTS 765 Table 1. Summary of Clinical Features of Cases 1-5 Case No. Age/sex Etiology of fistula Treatment Outcome 1 47/M CBD trauma at cholecystectomy 7F endoprosthesis Fistula closed in 3 days; stent removed after 8 wk 2 48/M Eroded cystic duct remnant 8F endoprosthesis Fistula closed in 4 days; stent removed with distal CBD stricture after 8 wk 3 78/F Cholangitis; subhepatic abscess 7F endoprosthesis; subsequent papillotomy Fistula closed in 2 days; stent removed after 20 wk 4 75/F Cholecystitis; CBD stricture; Papillotomy and 10F endoprosthesis Fistula closed in 4 days; stent left in place; patient died 7 mo later Fistula closed in 1 wk; stent removed after 4 wk probable pancreatic cancer 5 59/F Necrotizing pancreatitis with Percutaneous biliary stent abscess; persistent T-tube drainage CBD, common bile duct. drain. Six days later, abundant bile began to drain from the sump, and after this persisted for 1 mo, he was transferred to the MGH. After transfer, injection of contrast into the sump revealed free communication with the bile duct and dislodgement of the stent into the duodenum. After an unsuccessful attempt at percutaneous stent placement, endoscopic retrograde cholangiopancreatography was performed. Bile duct injection revealed extravasation of contrast from the bile duct just above the cystic duct (Figure 1A). A 7F double pigtail stent was positioned above the leak with the aid of a guide wire. Its distal end protruded from the ampulla (Figure 1B). Within 3 days the bile fistula closed. The sump drain was removed, and the patient was discharged 4 days after stent placement. Eight weeks later the stent was removed at repeat endoscopy. The patient remains well 11 mo later, and serum liver function tests are within normal limits. Case 2 A 48-yr-old man was admitted to the MGH for evaluation of 2 mo of nausea and episodic epigastric and back pain. After extensive evaluation, a diagnosis of ampullary stenosis was considered on the basis of provocative studies. Cholecystectomy, common duct exploration, and trans duodenal sphincterotomy were performed. At surgery the gallbladder contained no stones; the common duct exploration was negative, and an operative cholangiogram was normal. Following a normal T-tube cholangiogram on the eighth postoperative day, the T tube was removed. Subsequently, a Penrose drain site began to drain 500 mllday of bilious Figure 1. A. Endoscopic retrograde cholangiopancreatography showing extravasation of contrast material (arrow) from common hepatic duct just above the cystic duct (case 1). B. Overhead x-ray showing double pigtail stent in place bridging the site of extravasation (arrow) (case 1).

3 766 SMITH ET AL. GASTROENTEROLOGY Vol. 90, No.3 Figure 2. A. Endoscopic retrograde cholangiopancreatography showing extravasation of contrast from the cystic duct remnant (arrow) into a fistulous collection. The pancreatic duct is normal (case 2). B. Overhead x-ray showing 10F straight "Amsterdam" stent in place in the bile duct bridging the site of leakage (case 2). material. Copious bile drainage persisted for the next 2 wk. Injection of contrast material at the drain site revealed a complex right upper quadrant fistula tract to the biliary tree. Endoscopic retrograde cholangiopancreatography identified a point of extravasation from the cystic duct remnant (Figure 2A). No bile was seen to flow from the ampulla into the duodenum. An 8F straight "Amsterdam" endoprosthesis was placed across the site of extravasation using a guide wire (Figure 2B), and bile was immediately seen to flow into the duodenum. The bile leak ceased within 3 days, and the biliary-cutaneous fistula site closed and healed. Eight weeks later the stent was easily removed endoscopically. The patient has had no further problems in 10 mo of follow-up. Case 3 A 78-yr-old woman was referred to the MGH with fever, 6 mo of episodic right upper quadrant pain, nausea, and elevated serum glutamic oxaloacetic transaminase and serum alkaline phosphatase. She had undergone cholecystectomy 26 yr previously. Multiple unsuccessful attempts at transhepatic cholangiogram at the referring hospital were complicated by bleeding and spiking fevers. Endoscopic retrograde cholangiopancreatography revealed a dilated (20 mm) common bile duct without good visualization of the upper ducts. Laparotomy revealed a large bile-stained abscess encasing the porta hepatis and extending throughout the right upper quadrant and pararenal space. The abscess was drained, but the bile duct was not opened. Postoperatively, large amounts of bile leaked from the drains. The leak persisted without lessening for the next 2 wk. Repeat endoscopic retrograde cholangiopancreatography examination showed a 7-mm stone in the distal common bile duct, but the site of extravasation was not identified. A guide wire was passed to a point above the stone and a 7F double pigtail endoprosthesis was placed. Pus was seen to flow from the stent into the duodenum. After stent placement, the external bile drainage stopped within 48 h. The drains were removed from the abscess cavity, which healed promptly. Five months later the endoprosthesis was removed, and an endoscopic papillotomy was successfully completed. The patient remains well 5 mo later. Case 4 A 75-yr old woman was referred to the MGH because of a nonhealing biliary fistula. Her other medical problems included obesity, severe degenerative joint disease, osteoporosis with vertebral body fractures, and recurrent pulmonary emboli. For 3 yr she had complained of recurrent postprandial right upper quadrant pain. Ten weeks before referral she underwent cholecystectomy at an outside hospital for acute calculous cholecystitis, during which neither common bile duct exploration nor cholangiogram was performed. The postoperative course was complicated by a pulmonary embolus and a fever to 105 F. On the third postoperative day a biliary-cutaneous fistula developed through an abdominal drain site. The fistula failed to close during the next 2 mo. At transfer, aspartate transaminase, alkaline phosphatase, and bilirubin were modestly elevated. Endoscopic retrograde cholangiopancreatography revealed a normal ampulla. The common bile duct was strictured midway along its course and was dilated above the stricture. No stones were seen, nor was the extravasation site identified. A 1.0-cm papillotomy was performed, and a 10F Amsterdam stent was placed across the stricture. The nature of the stricture remained unclear. Carci-

4 March 1986 NONHEALING BCF WITH BILIARY STENTS 767 proach seemed to be contraindicated by the ongoing pancreatitis. the deformity of the duodenum due to the pancreatic phlegmon. and the recent abdominal surgery, a percutaneous approach to the bile duct was selected. Transhepatic cholangiography showed non dilated intrahepatic ducts, narrowing of the intrapancreatic portion of the common bile duct, and extravasation of contrast from the common bile duct just below the cystic duct remnant, presumably from the T-tube site. Drainage into the duodenum was achieved with an 8.3F multiple side-hole percutaneous catheter (Figure 3). Within 24 h the bile fistula drainage decreased to almost nil, and the abdominal wound began to granulate. Drainage through the fistula increased after 4 days, but this resolved completely after the catheter was exchanged for a 10F Argyle tube with no side holes near the site of extravasation. The wound healed rapidly and completely, and the biliary stent was removed after 1 mo. The fistula remains closed, and serum tests of liver function are normal 5 mo later. Figure 3. X-ray after transhepatic cholangiogram showing contrast leakage from the common bile duct below the cystic duct remnant (arrow). A percutaneous stent (arrowhead) has been placed in the common bile duct. bridging the site of leakage. Its pigtail end is seen in the duodenum (case 5). noma was suspected. but no mass was identified on abdominal computed tomography scan. Within 3 days after stent placement the fistula drainage had ceased. and the serum liver function tests had become normal. At the patient's request no further investigations were undertaken. She was discharged to her home feeling well but was readmitted to the referral hospital 7 mo later with jaundice and weight loss. At the family's request no further interventions were undertaken and the patient died. Permission for autopsy was refused. Case 5 A 59-yr-old white woman. 9 yr after cholecystectomy. presented to an outside hospital with sudden onset of severe epigastric pain and nausea. Necrotizing pancreatitis was discovered at emergency laparotomy. After common duct exploration. at which no stones were found. a drainage tube was left in the common bile duct. and a gastrostomy was performed. Recovery was complicated by fever. the development of a wound abscess. and leak of bile from the wound. The wound was opened and drained. and the T tube was removed. Profuse bile drainage (>900 mllday) continued for the next month while the wound deteriorated and enlarged progressively. On arrival at the MGH she had a 10 x 5-cm upper abdominal wound that extended down to the aorta at its base. A biliary-cutaneous fistula drained 1 Llday into the bed of the wound. After a computed tomography scan showed liquefaction necrosis of the tail of the pancreas. the dead tissues were surgically debrided and drained through a left-sided incision. Because an endoscopic ap- Discussion Biliary-cutaneous fistulas were reported in several series prior to 1900 (1,2). These cases occurred primarily in elderly patients with long histories of neglected gallbladder disease. Since 1900 fewer than 50 cases of spontaneous biliary-cutaneous fistulas have been reported; the improvement is probably due to better diagnosis and surgical therapy of stone-related biliary disease (3-5). Biliary leakage encountered in the modern era results mainly from inadvertent operative damage to the bile duct during cholecystectomy or common duct exploration, and may occur after uncomplicated operations on the biliary tree when strictures of the extrahepatic bile ducts cause increased bile duct pressures (6-8). The clinical manifestations of postoperative bile duct trauma include fever, jaundice, and biliary cutaneous fistula from the wound or drain sites. Though these complications are unusual, they represent difficult management problems. All our patients developed fistulas postoperatively-3 after cholecystectomy, 1 after T-tube placement, and 1 after multiple needle punctures of a biliary tree obstructed by a distal stone, subsequently complicated by a subhepatic abscess. Though a fistula may develop because of loss of bile duct wall without distal obstruction (case 1), such obstruction was the most common etiology of biliary-cutaneous fistula in our series (4 of 5 cases). The surgical drain site often provided the fistulous tract. Complications of chronic biliary-cutaneous fistula include steatorrhea, malabsorption of calcium and vitamin D, infection, and interference with wound healing. The latter was dramatically illustrated by case 5. Definitive treatment should be considered in a patient whose fistula persists for longer than 2 wk; in our patients, stent placement

5 768 SMITH ET AL. GASTROENTEROLOGY Vol. 90, No.3 was undertaken 2-8 wk after fistula formation (average, 4 wk). Standard therapy for nonhealing biliary-cutaneous fistula has previously been surgical, consisting of closure of the fistula, cholecystectomy (if not already done), and common bile duct exploration. Distal obstruction, the most common cause of fistula persistence, should be relieved when possible. Often, the fistula must be internalized by diversion into a Rnux-en-Y loop of jejunum. The operation is difficult and hazardous in such patients because of scarring and infection in the periportal region, and convalescence requires prolonged hospitalization. Adequate exposure to delineate the site of extravasation is often difficult, and though some estimates are higher, surgical mortality for such procedures is at least 3%-5% (9,10). Recent advances in interventional endoscopic and radiologic techniques offer a superior means of managing biliary-cutaneous fistulas. Percutaneous transhepatic cholangiography and biliary drainage have achieved widespread acceptance in the management of many complex biliary problems (11,12)' specifically for decompression of benign or malignant biliary obstruction. Recently, percutaneous transhepatic biliary drainage has been described in the management of a variety of biliary problems resulting from previous biliary operations (13). A subsequent series from the same group reports their success in 12 patients using percutaneous transhepatic biliary drainage for management of postoperative bile leaks and fistulas (14). Seven of their patients had bilomas or abscesses, and 5 patients had biliary-cutaneous fistulas. Two of the 12 patients required balloon dilatation of a bile duct stricture, but only 5 patients required definitive biliary tract surgery. Endoscopic sphincterotomy, first described 10 yr ago (15,16)' is now commonplace, with over 50,000 sphincterotomies performed worldwide (17-20). Though used primarily for bile duct stone disease, sphincterotomy has also been used in ampullary stenosis and ampullary neoplasms (21,22). O'Rahilly and colleagues (23) reported successful treatment with endoscopic papillotomy for a postoperative biliary-cutaneous fistula caused by choledocholithiasis. While in some cases sphincterotomy alone may be sufficient, use of a biliary stent serves two important functions in treating biliary-cutaneous fistulas. It provides relief of distal obstruction by providing a conduit, and bridges the hole at the site of bile extravasation. The biliary stent may also physically occlude the defect in the bile duct wall. Indwelling biliary stents can now be placed endoscopically through the ampulla of Vater over a guide wire (24,25). Early experience with the use of these stents in biliary obstruction is encouraging, although some patients develop recurrent jaundice or cholangitis due to stent occlusion or migration. When this occurs, the stent can be replaced quite easily; however, this is generally not a problem in managing biliary fistulas as the stents are usually a temporary measure. In our patients (except case 4), the stents were removed 4-20 wk after placement (average, 10 wk). The availability of equipment for placing larger diameter endoprostheses (up to 12F) allows increased bile flow; preliminary experience suggests longer stent patency and fewer occlusions. Placement of such stents requires a large duodenoscope with a mm biopsy channel and usually involves a preparatory papillotomy (26,27). A 10F Amsterdam stent was placed in patient 4 using this approach. Pigtail ends on biliary stents seem to offer no advantages for stent stability and reduce bile flow rates by about 50% (28). Hence, though clinical trials will be required to answer many questions about precisely the optimum stent characteristics, we are currently using 10F straight (Amsterdam) stent tubes. Percutaneous biliary stenting was performed in case 5 because pyloric and duodenal distortion from severe pancreatitis made the endoscopic approach undesirable. Immediately following percutaneous catheter placement the bile drainage ceased, and the large abdominal wound began to heal. The Johns Hopkins experience with percutaneous biliary drainage corroborates the validity of the stenting procedure in this entity, regardless of the route of introduction (13,14). The choice between the endoscopic or percutaneous approach to biliary fistula should depend on local expertise, the location of the site of extravasation, and the degree of bile duct dilation. Endoscopy offers the advantage of ampullary inspection, definitive therapy if papillotomy or stone extraction is required, and may be technically easier to perform than transhepatic drainage, especially in nondilated biliary ducts. In addition, successful internal drainage with an endoprosthesis is, in general, better tolerated by the patient than a percutaneous drain. References 1. Courvoisier 1. Pathologie and chirurgie der Gallenwege. Leipzig: FCW Vogel, Bonnet. Fistule biliaire cutanee. Lyon Med 1897;85: Henry CL, Orr TG Jr. Spontaneous external biliary fistulas. Surgery 1949;26: Hoffman L, Beaton H, Wantz G. Spontaneous cholecystocutaneous fistula: a complication of neglected biliary tract disease. J Am Geriatr Soc 1982;30: Ruderman RL, Laird W, Reingold MM, Rosen lb. External biliary fistula. Can Med Assoc J 1975;113: Warren KW, McDonald WM. Facts and fiction regarding strictures of the extrahepatic bile ducts. Ann Surg 1964; 159:

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