0016·5107/89/3506-0490$02.00 GASTROINTESTINAL ENDOSCOPY Copyright © 1989 by the American Society for Gastrointestinal Endoscopy
Endoscopic treatment of biliary tract fistulas Thierry Ponchon, MD, Jean-Franr;:ois Gallez, MD Pierre-Jean Valette, MD, Annick Chavaillon, MD Roger Bory, MD Lyon, France
Endoscopic therapy was attempted in 24 patients with spontaneous or postoperative persistent biliary fistulas. Endoscopic retrograde cholangiography demonstrated the site of the fistula in 22 cases. Sphincterotomy or biliary stent placement resulted in rapid resolution of the fistula in 16 of 24 patients. Failures were attributed to exclusion of the injured intrahepatic bile duct in two cases, insufficient dilation of a bile duct stricture in one, the large size of the bile duct defect in two, and associated lesions in three (cirrhosis, arterial trauma, sUbhepatic abcess). Endoscopic management of biliary fistulae requires: (1) visualization of the location of the fistula by retrograde cholangiography especially in case of an intrahepatic lesion, (2) prior percutaneous drainage of associated subhepatic or subphrenic abcesses, and (3) appropriate relief of distal biliary obstruction in order to reduce the intraductal biliary pressure. The outcome is uncertain when endoprostheses are used to bridge large bile duct defects. (Gastrointest Endosc 1989; 35:490-498)
The development of fistulas is a rare complication of biliary tract disease. They may occur as the result of neglected gallstones or as complications of peptic ulcer disease or hepatic hydatid cystS. 1- 3 The occurrence of these spontaneous fistulas has been dramatically reduced by the rapid diagnosis and treatment of these underlying conditions. Consequently, biliary fistulas are now usually seen following hepatobiliary surgery. Associated signs and symptoms include right upper quadrant pain, fever, and jaundice; they may also depend on the location of the fistula and include cutaneous biliary leakage, diarrhea, malabsorption, or manifestations due to biliary-pleural or biliary-bronchial fistulas. Surgery is usually necessary to relieve persistent biliary fistulas 4• 5 but is often difficult because of adhesions and associated inflammation. Reoperation for surgical injury of the biliary tract has a mortality ranging from 5 to 8%.6 Nonoperative alternatives have therefore been sought. Transhepatic treatment has been used7- 9 but has some disadvantages. The procedure may induce bleeding, the ampulla of Vater is not examined, and gallstone extraction can be hazardous. Furthermore, perReceived April 26, 1988. Forrevision August 10, 1988. Accepted April 27.1989. From Hepatogastroenterologie, Hopital Edouard Herriot. Lyon. Reprint requests: T. Ponchon, MD, Hepatogastroenterologie, Pavilion Hbis, Hopital E. Herriot, 69437 Lyon Cedex 03, France.
490
cutaneous transhepatic cholangiography is often difficult in patients with nondilated ducts, a frequent occurrence in biliary fistulas since the biliary system is decompressed. Recent advances in endoscopic techniques allow a new approach to the treatment of biliary fistulas. Biliary sphincterotomy,lO.l1 nasobiliary drainage, and the use of endoprosthesesl 2 '1 4 have all become routinely used techniques. Their application to the treatment of biliary fistulas has recently been described in a small number of patients. Thus, O'Rahilly et al. 15 reported the healing of a biliocutaneous fistula after endoscopic papillotomy and extraction of common bile duct stones. Burmeister et al. 16 reported a case of biliocutaneous fistula after surgery for hepatic trauma. The fistula closed within a few days after insertion of a nasobiliary drain. More recently, Sauerbruch et al. 17 reported four cases of post-operative biliary fistulas which were successfully treated by an endoscopically placed nasobiliary tube or an endoprosthesis. Smith et al. 18 also obtained excellent results after endoscopic insertion of biliary stents in four patients who had persistent heavily draining fistulas. Deviere et al. 19 reported closure of a post-traumatic biliary fistula by placement of a biliary stent. We report here our experience with endoscopic treatment in 24 patients with persistent biliary fistulas. GASTROINTESTINAL ENDOSCOPY
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No.
Cystectomy, cholecystectomy, and closure of a pleural fistula
Cystectomy and cholecystectomy, PD ofa postoperative subphrenic abcess Right hepatectomy, PD ofa postoperative subphrenic abcess -
PD of the gallbladder Surgical cholecystostomy
Hydatid cyst and cholelithiasis
Hydatid cyst and calculous gallbladder
Chronic calculous cholecystitis
Perforating acalculous cholecystitis Gangrenous calculous cholecystitis
Alveolar hydatid disease
Cholecystectomy and choledocotomy Left hepatectomy
Cholecystectomy, surgical injury (CBDO and HA)
Operative repair
Procedures
Cholelithiasis and choledocholithiasis Intrahepatic lithiasis
Traumatic liver and CBD injuries Gangrenous calculous cholecystitis
Initial disease
Table 1. Patients treated by sphincterotomy alone
Cholecysto-cutaneous viaPD Cholecysto-cutaneous via cholecystostomy
Cholecysto-colic
Hepatico-cutaneous via PD (undetected byERC)
Hepatico-cutaneous via PD
12
Hepatico-cutaneous via subphrenic drain Hepatico-bronchial and hepatico-cutaneous via subphrenic drain
60
10
-
7
10
8
7
25
30
Duration (days)
Choledochocutaneous via T-tube tract
Choledochocutaneous via T-tube tract Choledochocolic
Site of fistula
350
300
-
200
300
300
600
Cholangitis
Fever
Diarrhea
-
CBD stones
CBD stones
-
Hydatid debris in the CBD
-
ESand Dormia
ES
ESand Dormia
ES
ESand Dormia
ES
-
Fever and bilious sputum
ES
Papillary sclerosis
ES
ES
ES
Treatment
Papillary sclerosis
-
Distal biliary obstruction
-
Fever
Jaundice
Cholangitis and melena
-
300
Fever
Associated conditions
400
Volume (mil day) 3 days
3 days
2 days
3 days
2 days
Arterial bleeding after ES, persistent bile leak of 30 ml/day during 10 months Diarrhea stopped within 4 days, no extravasation on ERC 6 months after ES 3 days 1 day
Closure of cutaneous fistula in 3 days, closure of hepaticobronchial fistula on x-ray control 6 weeks after ES, relief of symptoms in 5 days 2 days
4 days
Relief of cholangitis in 5 days, persistence of hemobilia 10 days after ES, operative repair and choledochojejunostomy 5 days 9 days
Symptoms
Fistula
Duration posttreatment
PATIENTS AND METHODS Patients
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Between January 1980 and July 1987, 24 patients with persistent biliary fistulas were observed and treated by endoscopic methods (Tables 1 and 2). These patients included 17 men and 7 women aged 22 to 84 years (mean age, 57 years). Fistulas were spontaneous in one case, secondary to traumatic liver injury in two cases, and due to bile duct injury performed during hepatic, biliary, or gastric surgery in nine cases. The fistula occurred after hepatic surgery for hydatid disease in five cases and after apparently uncomplicated hepatic or biliary surgery in three patients. Fistulas were created by surgical cholecystostomy or percutaneous insertion of a catheter into the gallbladder under ultrasound guidance (percutaneous cholecystostomy) in four patients with gangrenous or perforating acute cholecystitis. Twenty-two patients presented with a persistent cutaneous bile leak. In six patients, bile was flowing through the surgical suction drains; in five, through a percutaneous catheter inserted under ultrasound guidance to drain a subphrenic or subhepatic abcess; in three, through a surgical cholecystostomy, and in one, through a percutaneous cholecystostomy. Fistulas followed the T-tube tract in four patients and the cystic drain tract in another. There were two fistulas draining through the operative cutaneous wound. The duration of the external bile leak before endoscopic treatment ranged from 7 to 80 days, and the quantity of biliary leakage ranged from 80 to 700 ml/day. Three patients had internal fistulas with the following symptoms: chronic diarrhea (case 8), melena following complex iatrogenic biliary trauma (case 2), and bilious sputum associated with an external fistula (case 5). Endoscopic methods
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Diagnostic endoscopic retrograde cholangiography (ERC) was first carried out in all patients using an Olympus JF1T or J3.7 duodenoscope. Sphincterotomy (Erlangen type sphincterotome, Olympus Optical Co. Ltd., Tokyo, Japan), nasobiliary drainage (Biotrol drain, Paris, France), or insertion of an endoprosthesis (Surgimed, Olstykke, Denmark) were then performed according to the results of ERC. Sphincterotomy alone was performed to extract calculi or hydatid debris, to treat papillary sclerosis, or to reduce the physiologic intrabiliary pressure in the absence of distal obstruction. Papillary sclerosis was diagnosed in patients presenting with a dilated common bile duct defined as a diameter in excess of 12 mm after correction for magnification and an irregular appearing sphincter of Oddi. Manometry was not available, and delayed contrast emptying through the sphincter of Oddi could not be assessed because of the fistula. Nasobiliary drainage (5 F gauge) was used to minimize intraductal biliary pressure. Stenting with an endoprosthesis (7 and 10 F gauge) was performed to dilate a benign stricture of the biliary tract or to bridge a large common bile duct injury. A bile duct injury was judged as "large" on cholangiogram when the biliary wall defect appeared to be circumferential or greater than 0.5 cm in length. Whenever possible, two prostheses were placed to avoid their dislodgement and to keep the stricture as widely dilated as possible. Antibiotics were administered after endoscopic procedures if sepsis (cholangitis, fever) was present. GASTROINTESTINAL ENDOSCOPY
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15
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Cholecystectomy, surgical CBD injury
Right hepatectomy
Procedures Hepaticocutaneous via subphrenic drain
Site of fistula
Choledochocutaneous via T-tube tract (large CBD defect on ERC) Acute calculous choCholecystectomy, Choledochocutaneous lecystitis surgical CBD invia cutaneous jury wound Perforating duodenal Billroth II proceCholedochocutaneous ulcer dure, surgical via cutaneous wound (large CBD CBD injury defect on ERC) Gangrenous calculous Surgical cholecysCholecystocutaneous cholecystitis tostomy via cholecystostomy Right hepatectomy, Choledochocutaneous Hepatic metastasis via PD (large CBD surgical CBD injury, PD of a defect on ERC) postoperative subhepatic abcess Hydatid alveolar dis- Right hepatectomy, Hepaticocutaneous surgical hilum via PD (large ease injury, PD of a LIHBD defect on postoperative ERC) subhepatic abcess Cholecystectomy, Choledochocutaneous Acute calculous chovia subhepatic surgical CBD inlecystitis drain (large CBD jury defect on ERC) Cholecystectomy, Choledochocutaneous Acute calculous chovia subhepatic surgical CBD inlecystitis drain (large CBD jury defect on ERC) Cholecystectomy, Choledochocutaneous Acute calculous chovia T-tube tract lecystitis surgical CBD injury Hydatid cyst Cystectomy, PD of Hepaticocutaneous a postoperative via PD (large subhepatic abRIHBD defect on cess ERC)
Traumatic liver injury
Initial disease
50/M Acute calculous cholecystitis
59/F
Age/ sex
14
13
No.
Table 2. Patients treated by nasobiliary drainage or insertion of endoprostheses
20
50
50
30
20
35
15
8
60
30
10
Duration (days)
Distal biliary obstruction Treatment
150
Fever
Cachexia
700
300
Septic shock 400
CBD stricture
-
3 days
7 days
4mo
4mo
4mo
4 mo
4 days
Persistent leak of 200 mlf day 50 days later, relief of fever in 5 days, operative repair
ES, two 10 FG stents
4 mo
No closure of fistula, death of hepatic failure and cachexia 2 days later
Persistent leak of 150 ml/ day 1 mo later, relief of sepsis in 7 days, operative repair
4 days
6 days
3 days
Persistent leak of 25 mlfday 1 mo later, relief of symptoms in 8 days, hepaticojejunostomy No closure of fistula and persistent sepsis 9 days later, operative drainage, death of sepsis 5 days 2 days 4mo
Fistula Symptoms Stenting
Duration post-treatment
ES, 10 FG stent
ES,1O FG stent
ES, two 10 FG stents
ES, 10 FG stent
-
200
-
ES, 10 FG stent
ES, Dormia, 10 FG stent
-
Cholangitis CBD stricture CBD stones
ES, two 7 FG stents
ES, two 10 FG stents
Cholangitis CBD stricture
-
ES, two 10 FG stents
Fever
Cholangitis LIHBD" stricture ES, 5 FG NBD for 3 weeks
Associated conditions
400
400
700
200
300
80
Volume (mlf day)
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The course of the disease after endoscopic treatment was evaluated by clinical examination, laboratory data (white blood cell count, serum total bilirubin, alkaline phosphatase, and aminotransferases), abdominal ultrasonography, and cystic duct cholangiography. Repeat ERe was performed to confirm closure of internal fistulas or to verify healing of the bile duct defect after removal of the endoprosthesis. RESULTS Radiographic data
Endoscopic retrograde cholangiography was successful in all cases. Extravasation of contrast material was noted in 22 patients. It originated from the gallbladder in 5 patients (cases 8 to 11 and 17), the cystic duct in 1 (case 24), an intrahepatic duct in 6 (cases 4 to 6, 13, 19, and 23) (Fig. 1), and the common bile duct in 10 (cases 1 to 3, 14 to 16, 18, and 20 to 22) (Fig. 2). The fistula tract was not detected by ERC in two patients (cases 7 and 12). The bile duct defect was judged as "large" in seven patients (cases 14, 16, 18 to 21, and 23). Three patients (cases 2, 3, and 11) presented with papillary stenosis. Benign common bile duct strictures occurred in four cases (nos. 15, 17, 22, and 24). A benign left intrahepatic duct stricture was present in one case (no. 13) and bile duct calculi or hydatid debris were present in four cases (nos. 6, 8, 10, and 17). One patient (case 17) had both a bile duct stricture and stones. In 13 patients there was no distal obstruction. Clinical results III
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Patients treated by sphincterotomy (cases 1 to 12). Nine patients were treated by sphincterotomy alone. The three others also required the use of a Dormia basket to remove calculi or hydatid debris. Nine patients were quickly cured by this method (Fig. 1). External fistulas disappeared within 2 to 5 days in eight patients. Resolution of internal fistulas was demonstrated by ERC in one patient and opacification via the cystic drain in another whose external fistula had also healed. Associated symptoms disappeared. Laboratory data returned to normal. Surgical drains and percutaneous catheters were easily removed. These nine patients were doing well with normal abdominal ultrasonography and normal laboratory data 6 to 12 months after the endoscopic procedure. Treatment failed in three patients. One patient, with a left intrahepatic bile duct injury, was operated on 6 days after an unsuccessful endoscopic procedure, and the left intrahepatic bile duct was found cut (case 12). Only the lower part of the intrahepatic bile duct had healed and the bile leak originated from the upper part of the injured bile duct and, therefore, was not visualized on ERC nor did it heal by sphincterotomy. In another patient (case 7) a small but persistent bile leak continued for 10 months after removal of the percutaneous GASTROINTESTINAL ENDOSCOPY
Figure 1. Cholangiogram via the cystic drain showing (A) post-operative hepatico-cutaneous (via the subphrenic drain) and hepatico-bronchial fistulas; (B) 6 weeks after sphincterotomy, closure of the fistulas (case 5).
catheter before subsequent spontaneous closure. In this case also, the fistula was undetected by ERC. A third patient (case 2) presented with persistent hemobilia 10 days after sphincterotomy. This patient was operated on because of an associated lesion of the hepatic artery and an arteriocholedocal fistula revealed by angiography. The initial cholangitis had resolved at the time of surgery. Patients treated by nasobiliary drainage and insertion of endoprostheses (cases 13 to 24). One patient (case 13) was treated with a 5 F gauge nasobiliary drain but underwent hepaticojejunostomy 1 month later because of a persistent external bile leak. Associated initial cholangitis had resolved at the time of surgery. Eleven patients were treated by insertion of endoprostheses (Fig. 2). Seven patients including four with a benign biliary stricture (cases 15, 17, 22, and 24) and three with a large bile duct injury (cases 16, 18, and 19) were cured by this technique (closure of the external fistulas within 1 to 7 days, followed by uncomplicated removal of the percutaneous catheter). Endoprostheses were removed after 4 months in these seven patients. Subsequent ERC demonstrated apparently normal or slightly enlarged bile ducts. Duration of follow-up after the removal of the endoprostheses ranged from 3 to 23 months. Subsequent physical examination and laboratory data have remained normal. VOLUME 35, NO.6, 1989
Insertion of endoprostheses failed to close the fistula in four patients with large bile duct defects (cases 14,20,21, and 23). One patient (case 21) with cirrhosis of the liver had a persistent biliary leak; he died of hepatic failure and cachexia 2 days after the endoscopic procedure. Three patients presented with fever (cases 14 and 23) or septic shock (case 20). Insertion of endoprostheses reduced but failed to stop the fistulous flow. Two patients (cases 20 and 23) then underwent successful surgical treatment of the fistula 30 and 50 days after the endoscopic procedure, by which time sepsis had disappeared. The third (case 14) underwent laparotomy 9 days after therapeutic endoscopy because of persistent sepsis. A subhepatic abcess was found and drained, and a choledochojejunostomy performed. The patient died of septic shock 5 days after surgery. The results according to the location of the biliary defect and causes of failure are summarized in Table 3. Fistulas originating from the gallbladder were successfully healed by endoscopic methods in five patients. Two of these patients (cases 9 and 10) underwent cholecystectomy 3 and 6 months later in order to prevent a septic recurrence. Three did not undergo cholecystectomy as they were high surgical risks. Complications
Bleeding at the site of sphincterotomy was observed in one patient, but stopped within 48 hours after one 495
DISCUSSION
2. (A) After a right hepatectomy for hydatid disease, a large defect of the hilum and the proximal part of the left intrahepatic bile duct is demonstrated (arrows) by a temporarily placed 7 F gauge nasobiliary drain. (8) A 10 F gauge endoprosthesis is inserted in the left intrahepatic bile duct in order to bridge the defect. Closure of fistula was obtained in 4 days. (C) Immediately after removal of the endoprosthesis, bile duct is found slightly enlarged at the site of fistula (arrows). Eleven months later, ultrasonographic and biochemical data were normal (case 19).
Figure
session of laser photocoagulation. No serious complication due to nasobiliary drainage or the use of endoprostheses was noted. 496
Endoscopic techniques offer a new and effective approach to the treatment of biliary fistulas. In 16 of our 24 patients, endoscopic treatment resulted in prompt resolution. Four factors should be taken into account during endoscopic procedures in this type of patient: the type of distal biliary obstruction, the size of the bile duct injury, the location of the injury, and the associated lesions or general condition of the patient. Type of distal biliary obstruction. The decision to use sphincterotomy alone or to insert an endoprosthesis after sphincterotomy depends on the type of distal biliary obstruction including papillary sclerosis, bile duct stricture, stones, or hydatid debris in the common bile duct. It should be emphasized that sphincterotomy may even be considered if no distal obstruction is demonstrated on cholangiography. Under physiologic conditions there is a 10 mm Hg pressure gradient between the biliary system and the duodenum. 2o Sphincterotomy may lead to a sufficient reduction in the normal biliary tract pressure, resulting in closure of the fistula. This was achieved in four of six patients. Spontaneous healing of the fistulas could have been expected. However, endoscopic treatment was indicated because of the large volume of bile leakage (200 to 600 ml/day) and the associated symptoms (fever, bilious sputum). Three patients were diagnosed with papillary sclerosis on cholangiography, but manometry was not performed. Nevertheless, sphincterotomy was also used in these cases to reduce the gradient of pressure between the biliary tree and the duodenum. In theory, a nasobiliary drain is complementary to sphincterotomy allowing for more effective lowering of the intraductal pressure; it also permits contrast infusion at a later date for a follow-up cholangiogram. However, it is uncomfortable for the patient and may prolong metabolic acidosis secondary to bile loss. In our series, nasobiliary drainage was obviated because of the rapid response of the fistulas to sphincterotomy alone. In the only case where a 5 F nasobiliary drain was used, the indication was not optimal as a biliary stricture was present. Concerning the use of endoprostheses in cases of bile duct strictures, the optimum duration of stenting is not clear. Large gauge (10 F) stents have reduced the risk of blockage by debris. We left the stents in place for 4 months while the fistulas closed, and the cholangiographic picture improved. Huilbregtse et al. 21 advocate replacement every 3 months if possible and removal after 6 months if the patient is doing well and if strictures appear adequately dilated. In our study, the limited follow-up and the small number of patients does not permit us to conclude whether two or even GASTROINTESTINAL ENDOSCOPY
Table 3. Summary of clinical results according to the location of the biliary defect Common bile duct (10)" I
Success (6)
I
I
Failures (4)
Intrahepatic bile duct (8) I
I
Success (4)
I I I Associated arteriocholedochal fistula
(1)
Large choledochal defect (1) Associated subhepatic abscess (1) Poor general condition (1)
I
Failures (4)
Gallbladder (5)
Cystic duct (1)
,--l---,
,----L-,
Success (5)
Success (1)
I I Insufficient dilation of associated LIHBD b stenosi3 (1) Large RIHBD defect (1) No detection of biliary tract defect on ERC (2)
" Numbers in parentheses, number of cases. b LIHBD, left intrahepatic bile duct; RIHBD, right intrahepatic bile duct; ERC, endoscopic retrograde cholangiography.
three endoprostheses are more effective than one, and whether strictures recur after removal of the endoprostheses. Size of the bile duct injury. In our opinion, endoprotheses can often be used to bridge a large bile duct defect because complications are uncommon. But the results are not conclusive: only 3 of 7 patients were completely healed by this technique in our series, and the follow-up period was too brief to ascertain that no secondary bile duct stricture was present. Location of the bile duct injury. Fistulas originating in the gallbladder, with or without cholelithiasis, or in the cystic duct, seem to close rapidly with endoscopic treatment. Resolution of fistulas originating from the common bile duct depends on associated lesions and the size of the bile duct defect. Intrahepatic bile duct injuries are more difficult to manage. First, intrahepatic strictures and defects are not easily accessible by the retrograde route. Second, their etiology is not always clear and the injury may be complex. The lower part of the injured bile duct may be closed and ERC, therefore, may fail to reveal any contrast extravasation. Bile can continue to leak from the upper part of the injury, and this will not respond to endoscopic treatment. It is now quite clear to us that endoscopic treatment is likely to be without value when ERC does not reveal the fistula. Associated lesions. In two cases, failure was unrelated to the biliary fistula itself. It was due to an associated arterial lesion in one patient and to an underlying poor general condition with hepatic failure in another. These cases are not a reflection on the efficacy of the endoscopic procedure. On the other hand, it is much more important to take into consideration the presence of associated subhepatic or subphrenic abscesses. If these abscesses are poorly connected or not connected to the tract of the fistula, drainage of the bile ducts by retrograde route cannot cure them. In our series, abcesses were drained by a catheter inserted percutaneously under ultrasound guidance before the endoscopic procedure in five patients. In another patient (case 14), a subhepatic abVOLUME 35, NO.6, 1989
cess was overlooked and sepsis continued after endoscopic treatment. Subsequent surgery allowed drainage but was probably performed too late, and the patient died from septic shock. In the case of sepsis associated with a biliary fistula, abscesses have to be carefully searched for, not only on cholangiography and fistulography but principally on ultrasonography and CT scanning, especially if endoscopic treatment is being considered. If an abscess is detected, two therapeutic procedures are possible: (1) endoscopic drainage of bile ducts associated with percutaneous drainage of the abscess or (2) early surgery. We prefer the first solution, if possible, because of its low morbidity; but our experience includes only five cases. Of course, if the abscess is too large to be drained by the percutaneous route or if there is peritonitis, early surgery is the only solution. Endoscopic treatment should be considered as the therapeutic method of choice in patients with biliary fistulas. It can provide a rapid and safe treatment for these often critically ill patients. It requires: previous visualization of the location of the fistula by retrograde cholangiography, especially in cases of intrahepatic lesions; previous detection and percutaneous drainage of associated subhepatic or subphrenic abscesses; and appropriate relief of distal biliary obstruction in order to reduce the intraductal biliary pressure. The outcome is uncertain when endoprostheses are used to bridge large bile duct defects. ACKNOWLEDGMENTS
Our thanks to Drs. S. Sidi, E. Krawitt, and A. Barkun for reviewing the manuscript.
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GASTROINTESTINAL ENDOSCOPY