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Biliary intestinal fistula
Biliary Intestinal Fistula JAMES R. STULL, MD, Columbus, Ohio NElL R. THOMFORD, MD, Columbus, Ohio
Because biliary intestinal fistulas have no specific ~yrnptoms or physical signs, they are often overlooked mtil related complications occur. These complications it~clude gallstone iteus, obstruction of the common bile duct, cholangitis, hemorrhage from the fistula, :,nd obstruction of the small intestine or colon as a ~csult of extrinsic inflammation. The management of i~atients with biliary intestinal fistulas is therefore ~sually complex and often controversial. Whether or ,aot the fistula should be repaired during operations for gallstone ileus and what procedure to perform in cases in which a fistula is the result of peptic ulcer :~re examples of the problems in management. This p~per is a review and discussion of the etiology, associated complications, and management of twentytwo patients with biliary intestinal fistulas.
Clinical Material The case records of all patients with biliary fistulas admitted to The Ohio State University Hospitals in the twenty year period from July 1, 1949 through June 30, 1968 were reviewed. A total of twenty-two cases were recognized in which there was the spontaneous development of a fistula between the gallbladder or bile ducts and the small or large intestine:. (Table I.) Tilere were thirteen female and nine male patient~ whose ages ranged from fifty-two to eighty-four years. Of the twenty-two cases, fifteen fistulas were cholecystoduodenal, six were cholecystocolonic, and one was choledochoduodenal. Fistulas resulted from disease ~f the gallbladder in nineteen cases, duodenal ulcer i~1 two cases, and recurrent pancreatitis in a single ,.:ase.
Eight of the twenty-two patients presented with .:lassical symptoms and physical signs of chronic chole,:ystitis including intermittent episodes of pain in the ~ight upper quadrant of the abdomen. Six patients !~resented with symptoms and signs of mechanical ,:bstruction of the intestine; in four of these cases the ~bstruction was caused by a gallstone. A marked in:tammatory reaction associated with development of rein the Department of Surgery, The Ohio State University College ~f Medicine, Columbus, Ohio. Reprint requests should be addressed to Dr Thomford. 410 West tenth Avenue, Columbus, Ohio 43210.
Vol. 120, July 1970
the fistula resulted in mechanical obstruction of the ascending colon of one patient and the second portion of the duodenum in another. One patient was admitted because of a duodenal ulcer and uremia. Two patients were hospitalized because of intermittent episodes of fever, chills, and jaundice, caused by ascending cholangitis. One patient with a fistula between the gallbladder and colon complained of diarrhea and hematochezia. Tile causes of hospitaliTation in the remaining patients were cecal polyp, adenocarcinoma of tile colon, neuromuscular dystrophy, and uremia secondary to chronic pyeloncphritis, each in a single case. In only thirteen of the twenty-two patients did questioning concerning their past medical history suggest symptoms of disease of the gallbladder or bile ducts. Two of the thirteen patients had undergone cholecystostomy. The ten patients with symptoms of chronic chole.. cystitis or ascending cholangitis had varying degrees of tenderness to palpation in the epigastrium and right upper quadrant of the abdomen. Ti~e six patients with mechanical obstruction of the intestine had mild to moderate distention of the abdomen and peristaltic sounds typical of mechanical obstruction. None of the twenty-two patients had a palpable mass in the right upper quadrant of the abdomen or hepatomegaly. The serum bilirubin was normal in twelve of eighteen patients tested; in four patients elevations were less than 2.0 mg per cent, and in the remaining two patients the recorded values were 4.3 and 11.2 mg per cent, respectively. Anemia caused by blood loss from the fistula occurred in only one case. In cases of cholecystoduodenal fistulas plain roentgenograms of the abdomen showed air in the biliary tree in eight (57 per cent) of foune~.n patients examined; contrast media was seen in the biliary tree of nine (69 per cent) of thirteen patients in whom roentgenograms were taken after a meal of barium sulfate. The six patients with cholecystocolonic fistulas were all examined with plain roenlgenograms of the abdomen and in five of the six patients, rocntgenograms were made after an enema of barium sulfate; air in the biliary tree and contrast media in the biliary tree were seen in only a single patient each. In the single case of choledochoduodenal fistula, roentgenograms
27
Stull and Thomford
TABLE I
Billary Intestinal Fistulas in Twenty-Two Patients
Case Age (yr) Number and Sex
28
Fistula
Associated Complications
PreviousRelated Operations
Operation
1
84, F
Cholecystoduodenal
Cholangttis
None
2
43, F
Cholecystoduodenal
None
None
3
5g, F
Cholecystoduodenal
None
4
75, F
Cholecystoduodenal
Gallstone ileus
Removal of gallstone obstructing ileum None
5
68, F
Cholecystoduodenal
None
Cholecystostomy; choledochostomy
6
64, M Cholecysteduodenal
None
None
7
74, F
Cholecystoduodenal
None
8
70, F
Cholecystoduodenal
None
9
55, F
Cholecystoduodenal
Gallstone obstructing duodenum Gallstone obstructing duodenum None
Cholecystectomy, closure of duodenum Cholecystectomy; closure of duodenum Cholecystectomy; closure of duodenum Cholecystectomy; closure of duodenum; enterolithotomy Cholecystectomy; choledocholithotomy; closure of duodenum Cholecystectomy; closure of duodenum Cholecystectomy; closure of duodenum; enterolithotomy Gastrojejunostomy
None
None
10
52, F
Cholecystoduodenat
Non~
None
None
11
60, F
Cholecystoduodenal
None
None
None
12
57, F
Cholecystoduodenal
None
None
None
13
62, F
Cholecystoduodenal
Gallstone ileus
None
None
14
78, F
Cholecystoduodenal
Cholangitis
None
None
15
83, M Cholecystoduodenal
Cholangitis
None
None
16
68, F
Cholecystocolonic
None
None
17
65, F
Cholecystocolonic
Hone
None
18
63, M Cholecystocoionic
19
77, M Cholecystocolonlc
Cholecystostomy; right hemicolectomy Cholecystectomy; closure of colon, choledocholithotomy Cholecystectomy; closure of colon Cecostomy
21
55, F
22
75, M Choledochoduodenal
Cholecystocolonic
Hematochezia for None 24 hr Mechanical None obstruction of ascending colon by extrinsic inflammation None Cholecystostomy Chronic pancreatitis
None
Cholecystectomy; closure of colon; choledocholithotomy None
Resultand Comments Recovered Recovered Recovered Recovered Recovered
Recovered Recovered
Died in 2 mo of aplastic anemia Died in 3 wk of thymoma Fistula asymptomatic; multiple myeloma Died in 5 mo of renal failure Metastatic adenocarcinoma of colon Passed 3 cm stone per rectum; asymptomatic 10 yr later Asymptomatic 7 yr later Lost to follow-up study Adenomatous polyps of cecum Recovered
Recovered Cholecystectomy; right hemicolectomy in 3 wk
Recovered
Lost to follow-up study
The American Journal of Sul'ger~,
Biliary Intestinal Fistula before and after a meal of barium sulfate showed contrast media bat no air in the biliary tree. Fourteen of the twenty-two patients underwent operation. Operations were performed in eight of the ~ifteen patients with cholecystoduodenal fistula; two of daese patients had gallstone ileus and were managed !~y removal of the obstructing stone in one patient and ~emoval of the stone, cholecystectomy, and closure of ~he duodenum in the other. Five of the eight patients mderwent cholecystcctomy and closure of the duo,~cnum. A gastroenterostomy was established in the :emaining patient because of mechanical obstruction ~,f the duodenum caused by the inflammation as....~ciated with the cholecystoduodenal fistula. All eight ~,atients recovered. The patient with gallstone ileus, :nanaged initially by only the removal of the obstruct:ag stone, subsequently underwent cholccystectomy ~nd closure of the duodenum. Of the eight patients Mth ¢holecystoduodenal fistula who underwent opera~ion, stones were removed from the common bile duct ~f one patient and operative cholangiography was reported as showing nothing abnormal in a second patient. In the six remaining patients the common bile duct was not opened at the time of operation. One of the six patients had an episode of chills, fever, and jaundice one month after operation, and postmortem examination of another patient who later died of aplastic anemia revealed calculi within the common bile duct. Operation was not performed in four patients with cholecystoduodenal fistula because of metastalic adenocarcinoma, terminal uremia, neuromuscular dystrophy, and multiple myeloma each in a single case. Three patients with cholecystoducxtenal fistula refused operation; two had severe cholangitis and the third had gallstone ileus. All three patients recovered; two t~f the three patients passed gallstones per rectum. One of the three patients was lost to follow-up study; ~hc other two have been asymptomatic for seven and ~'cn yeats, respectively. Operations were performed in all six patients with cholccystocolonic fistula. Four of the six underwent cholecystectomy and closure of the defect in the ;~cending colon. Cholecystectomy and right hemicolec~'~lny were performed in the patient with associated ;,olyps of the cecum. The patient who presented with :~cchanical obstruction of the ascending colon initially underwent cecostomy followed after one month v cholecystectomy and removal of a segment of the :~cending colon. Cholcdochotomy was performed in ~hrec of the six patients with cholecystocolonic fistula, ::nd calculi were found in the common bile duct in ..ach case. All six patients recovered after operation. The single patient with a choledochoduodenal fistula, seventy-five year old man with symptoms and signs
voL 120, July ]970
of recurrent pancreatitis, improved with medical management, was discharged from the hospital, and unfortunately was lost to follow-up study. Comments The relative incidence of each type of internal biliary fistula was demonstrated by Waggoner and Le Mone's review [I] of 819 eases in which they ftmnd 51 per cent were cholecystoduodenal, 21 per cent cholecysto=olonic, and 19 per cent choledochoduodenal. The remaining cases included cholecystogastric, choledochogastric, and cholecystocholedochal fistulas, in rare instances biliary fistulas have also been reported to extend into the renal pelvis, uterus, p~rtal vein, pericardium, and bronchial tree [2]. When all types of internal biliary fistulas are totaled, the over-all incidence is reported as 0.l to 0.5 per cent in autopsy series and 1.2 to 5.0 per cent in large series of operations to remove the gallbladder [2--4]. The fact that biliary-entefic listula will heal st~ntaneously may explain the lower incidence in autopsy studies. Some internal biliary fistulas are the result of operative or other trauma, but most occur spontaneously. Those fistulas which occur spontaneously between the gallbladder or bile ducts and the intestine are usually the result of chronic cholecystitis, peptic ulcer, or neoplasm. Since in most instances the etiology and problems in management of intestinal fistulas from the gallbladder differ from those of fistulas of the bile ducts, they should be considered and discussed as separate entities. The development of fistulous tracts from the gallbladder is associated with gallstones in as many as 90 per cent of cases [5]. Initially, adhesions between the gallbladder and the intestine are produced by recurrent attacks of cholecystitis. When inflammation and pressure of calculi cause necrosis of the wall of the gallbladder, the adherent portion of the intestine is destroyed in a similar manner and a fistula is formed. Because of its proximity, the duodenum is the most common portion of the intestine involved. As might be expected, the hepatic flexure of the colon is the next most frequent site of a fistula from disease of the gallbladder. Fistulas between the bile ducts and lhe duodenum, in contrast to those between the gallbladder and duodenum, are usually the result of peptic ulceration of the duodenum. As many as 80 per cent of choledochoduodenal fistulas are caused by penetrating duodenal ulcers which erode the anterior wall of the common bile duct [6]. Only in rare instances d o peplic ulcers cause a fistula between the duodenum and the gallbladder. In one series of thirty-six patients with biliary intestinal fistula secondary to peptic ulcer, thirty fistulas
29
Stull and Thomford were choledochoduodenal whereas only six were choleeystoduodenal [7]. There are no specific symptoms which suggest the development or presence of a biliary intestinal fistula. In some instances a fistula from the gallbladder into the duodenum may improve the condition of the patient by decompressing an obstructed and inflamed gallbladder. Symptoms, if any, from a choledochoduodenal fistula caused by peptic ulceration are usually overshadowed by the chronic recurrent symptoms of the ulcer. Exceptions to the common paucity of symptoms are the development of gallstone ileus, or, in a patient with cholecystocolonic fistula, the onset of diarrhea. Of the twenty-two patients described in this report, four had symptoms and signs of gallstone ileus. Only one of six patients with cholecystocolonic fistula had noted significant diarrhea. There are no studies of blood, serum, or urine which are diagnostic of biliary intestinal fistula, and identification of a fistula by cholecystography or cholangiography is rare. In contrast, roentgenograms of the abdomen both before and after a meal of barium are often diagnostic. In this series, air was seen in the biliary tree in 57 per cent of p~tients who had plain roentgenograms of the abdomen. Rocntgenograms after barium studies showed barium in the biliary tree in 69 per cent of the patients examined. When evaluating the individual case, however, it is important to recall that air in the biliary tree is not pathognomonic of a fistula [8]. In rare instances air in the bile ducts may be seen as a result of an incompetent sphincter of Oddi. Complications of biliary intestinal fistulas include gallstone ileus, ascending cholangitis, severe diarrhea in cases of cholecystocolenic fistula, and gastrointestinal hemorrhage [9,10]. Gallstone ileus is of particular concern since the mortality of this complication has been reported to be as great as 50 per cent in some series. Obstruction of the intestine by a gallstone, usually lodged in the terminal portion of the ileum, accounts for 1 to 5 per cent of all cases of intestinal obstructio~t. Most authors have advocated removal of the obstructing calculus at the initial operation with repair of the fistula at a later date. There have been exceptions to this rule [9]. The decision to repair the fistula in addition to relieving the intestinal obstruction should be an intraoperative one determined by the general condition of the patient, the characteristics of the inflammatory process surrounding the fistula, and the extent of the operation necessary to divide the fistulous tract, correct the underlying disease, and repair the involved organs. Although in most instances hemobilia is the result of rupture of an aneurysm of the cystic or hepatic
30
artery, a few cases of hemorrhage from biliary duodenal fistula have been reported [9,10]. Hemorrh,'tge from a cholecystocolonic fistula is more rare with only seven such cases having been reported [lO]. In at least one instance of cholecystocolonic fistula, severe hemorrhage occurred after erosion of the cystic artery. Operations for cholecystoduodenal and cholecystocolonic fistulas caused by inflammatory disease of the biliary tree are relatively standard when there are no related complications. Cholecystectomy should be performed and the colon or duodenum separated from the biliary tree and repaired. In selected cases, choleeystostomy rather than cholecystectomy may be necessary because of severe inflammation. In other instances, such as when a mass within the colon or gallbladder suggests that the fistula is the result of malignant disease, en bloc removal of the adjacent tissues may be appropriate. When biliary intestinal fistulas are caused by peptic ulceration, primary attention must be given to correction of the ulcer diathesis which caused the complication. When it is technically possible, hemigastrectomy, closure of the duodenum, vagotomy, and gastrojejunostomy are advisable. This provides acceptable management of the ulcer and excludes the area of the fistula from the gastrointestinal stream. Any additional procedures on the biliary tree or fistula at the initial operation will depend on the general condition of the patient and the intraoperative findings. Obstruction to the flow of bile must be alleviated if only by catheter drainage of the bile ducts. Ideally, the fistula should be divided and the duodenum repaired together with appropriate correction of disease or defects in the biliary tree. In some instances inflammation may have resulted in fibrosis with a permanent stricture of the common bile duct. These cases may require choledochoduodenostomy or choledochojcjunostomy. It is important to emphasize that in all cases of biliary intestinal fistula, the anatomy of the fistula and the presence or absence of calculi or other abnormalities within the biliary tree should be carefully assessed by intraoperative cholangiography.
Summary Biliary intestinal fistulas developed spontaneously in twenty-two patients; fifteen of the fistulas were cholecystoduodenal, six were cholecystocolonic, and one was choledochoduodenal. Fourteen of the twentytwo patients underwent an operation for the fistula or a related complication; there were no postoperative deaths. Fistulas from the gallbladder to the duodenum are most common and usually result from chronic cholecystitis. Fistulas of the common bile duct are in The American Journal Of Surgery
Biliary Intestinal Fistula most cases due to a peptic ulcer; with the exception L,f life-threatening complications, the primary goal in tt~cse cases is correction of the ulcer diatheses. References 1. Waggoner CM, LeMone DV: Clinical and roentgen aspects of internal biliary fistulas, Radiology 53: 31. 1949. 2. Puestow CB: Spontaneous internal biliary fistula. Ann Sur8 115: 1043. 1942. 3. Noskin EA, Strauss AA. Strauss SF: Spontaneous in. ternal biliary fistula: a review of the literature and report of two cases, Ann Surg 130: 270, ]949. 4. Pitman RG, Davies A: The clinical and radiological features of spontaneous internal biliary fistulae. Brit J Surg 50: 414, 1962-3.
V,~I, 120, July I970
5. HIcken N, Coray QB: Spontaneous gastrointestinal biltary fistulas. Surg Gynec Obstet 82: 723, ]946. 6. Hutchings VC, Wheeler JR. Puestow CB: Choledocho, duodenal fistula complicating duodenal ulcer. Arch Surg 73: 598, 1956. 7. Kourias GB, Choultaras A: Spontaneous gastrointestinal billary fistula complicating duodenal ulcer. Surg Gynec Obstet 119: 1013, 1964, 8. Constant E0 Turcotte JG: Choledochoduodenal fistula: the natural history and management of an unusual complication of peptic ulcer disease. Ann Surg 167: 220, 1968. 9. Carlson EG, Gates CY, Novacovich G; Spontaneous fis. tulas between the 8allbladder and gastrointestinal tract. Surg Gynec Obstet 101: 321, 1955. 10. Kaplan BJ: Massive lower 8astrointestinal hemorrhage from cholecystocolic fistula. Dis Colon Rectum 10: 191, 1967,
31
Because biliary intestinal fistulas have no specific ~yrnptoms or physical signs, they are often overlooked mtil related complications occur. These complications it~clude gallstone iteus, obstruction of the common bile duct, cholangitis, hemorrhage from the fistula, :,nd obstruction of the small intestine or colon as a ~csult of extrinsic inflammation. The management of i~atients with biliary intestinal fistulas is therefore ~sually complex and often controversial. Whether or ,aot the fistula should be repaired during operations for gallstone ileus and what procedure to perform in cases in which a fistula is the result of peptic ulcer :~re examples of the problems in management. This p~per is a review and discussion of the etiology, associated complications, and management of twentytwo patients with biliary intestinal fistulas.
Clinical Material The case records of all patients with biliary fistulas admitted to The Ohio State University Hospitals in the twenty year period from July 1, 1949 through June 30, 1968 were reviewed. A total of twenty-two cases were recognized in which there was the spontaneous development of a fistula between the gallbladder or bile ducts and the small or large intestine:. (Table I.) Tilere were thirteen female and nine male patient~ whose ages ranged from fifty-two to eighty-four years. Of the twenty-two cases, fifteen fistulas were cholecystoduodenal, six were cholecystocolonic, and one was choledochoduodenal. Fistulas resulted from disease ~f the gallbladder in nineteen cases, duodenal ulcer i~1 two cases, and recurrent pancreatitis in a single ,.:ase.
Eight of the twenty-two patients presented with .:lassical symptoms and physical signs of chronic chole,:ystitis including intermittent episodes of pain in the ~ight upper quadrant of the abdomen. Six patients !~resented with symptoms and signs of mechanical ,:bstruction of the intestine; in four of these cases the ~bstruction was caused by a gallstone. A marked in:tammatory reaction associated with development of rein the Department of Surgery, The Ohio State University College ~f Medicine, Columbus, Ohio. Reprint requests should be addressed to Dr Thomford. 410 West tenth Avenue, Columbus, Ohio 43210.
Vol. 120, July 1970
the fistula resulted in mechanical obstruction of the ascending colon of one patient and the second portion of the duodenum in another. One patient was admitted because of a duodenal ulcer and uremia. Two patients were hospitalized because of intermittent episodes of fever, chills, and jaundice, caused by ascending cholangitis. One patient with a fistula between the gallbladder and colon complained of diarrhea and hematochezia. Tile causes of hospitaliTation in the remaining patients were cecal polyp, adenocarcinoma of tile colon, neuromuscular dystrophy, and uremia secondary to chronic pyeloncphritis, each in a single case. In only thirteen of the twenty-two patients did questioning concerning their past medical history suggest symptoms of disease of the gallbladder or bile ducts. Two of the thirteen patients had undergone cholecystostomy. The ten patients with symptoms of chronic chole.. cystitis or ascending cholangitis had varying degrees of tenderness to palpation in the epigastrium and right upper quadrant of the abdomen. Ti~e six patients with mechanical obstruction of the intestine had mild to moderate distention of the abdomen and peristaltic sounds typical of mechanical obstruction. None of the twenty-two patients had a palpable mass in the right upper quadrant of the abdomen or hepatomegaly. The serum bilirubin was normal in twelve of eighteen patients tested; in four patients elevations were less than 2.0 mg per cent, and in the remaining two patients the recorded values were 4.3 and 11.2 mg per cent, respectively. Anemia caused by blood loss from the fistula occurred in only one case. In cases of cholecystoduodenal fistulas plain roentgenograms of the abdomen showed air in the biliary tree in eight (57 per cent) of foune~.n patients examined; contrast media was seen in the biliary tree of nine (69 per cent) of thirteen patients in whom roentgenograms were taken after a meal of barium sulfate. The six patients with cholecystocolonic fistulas were all examined with plain roenlgenograms of the abdomen and in five of the six patients, rocntgenograms were made after an enema of barium sulfate; air in the biliary tree and contrast media in the biliary tree were seen in only a single patient each. In the single case of choledochoduodenal fistula, roentgenograms
27
Stull and Thomford
TABLE I
Billary Intestinal Fistulas in Twenty-Two Patients
Case Age (yr) Number and Sex
28
Fistula
Associated Complications
PreviousRelated Operations
Operation
1
84, F
Cholecystoduodenal
Cholangttis
None
2
43, F
Cholecystoduodenal
None
None
3
5g, F
Cholecystoduodenal
None
4
75, F
Cholecystoduodenal
Gallstone ileus
Removal of gallstone obstructing ileum None
5
68, F
Cholecystoduodenal
None
Cholecystostomy; choledochostomy
6
64, M Cholecysteduodenal
None
None
7
74, F
Cholecystoduodenal
None
8
70, F
Cholecystoduodenal
None
9
55, F
Cholecystoduodenal
Gallstone obstructing duodenum Gallstone obstructing duodenum None
Cholecystectomy, closure of duodenum Cholecystectomy; closure of duodenum Cholecystectomy; closure of duodenum Cholecystectomy; closure of duodenum; enterolithotomy Cholecystectomy; choledocholithotomy; closure of duodenum Cholecystectomy; closure of duodenum Cholecystectomy; closure of duodenum; enterolithotomy Gastrojejunostomy
None
None
10
52, F
Cholecystoduodenat
Non~
None
None
11
60, F
Cholecystoduodenal
None
None
None
12
57, F
Cholecystoduodenal
None
None
None
13
62, F
Cholecystoduodenal
Gallstone ileus
None
None
14
78, F
Cholecystoduodenal
Cholangitis
None
None
15
83, M Cholecystoduodenal
Cholangitis
None
None
16
68, F
Cholecystocolonic
None
None
17
65, F
Cholecystocolonic
Hone
None
18
63, M Cholecystocoionic
19
77, M Cholecystocolonlc
Cholecystostomy; right hemicolectomy Cholecystectomy; closure of colon, choledocholithotomy Cholecystectomy; closure of colon Cecostomy
21
55, F
22
75, M Choledochoduodenal
Cholecystocolonic
Hematochezia for None 24 hr Mechanical None obstruction of ascending colon by extrinsic inflammation None Cholecystostomy Chronic pancreatitis
None
Cholecystectomy; closure of colon; choledocholithotomy None
Resultand Comments Recovered Recovered Recovered Recovered Recovered
Recovered Recovered
Died in 2 mo of aplastic anemia Died in 3 wk of thymoma Fistula asymptomatic; multiple myeloma Died in 5 mo of renal failure Metastatic adenocarcinoma of colon Passed 3 cm stone per rectum; asymptomatic 10 yr later Asymptomatic 7 yr later Lost to follow-up study Adenomatous polyps of cecum Recovered
Recovered Cholecystectomy; right hemicolectomy in 3 wk
Recovered
Lost to follow-up study
The American Journal of Sul'ger~,
Biliary Intestinal Fistula before and after a meal of barium sulfate showed contrast media bat no air in the biliary tree. Fourteen of the twenty-two patients underwent operation. Operations were performed in eight of the ~ifteen patients with cholecystoduodenal fistula; two of daese patients had gallstone ileus and were managed !~y removal of the obstructing stone in one patient and ~emoval of the stone, cholecystectomy, and closure of ~he duodenum in the other. Five of the eight patients mderwent cholecystcctomy and closure of the duo,~cnum. A gastroenterostomy was established in the :emaining patient because of mechanical obstruction ~,f the duodenum caused by the inflammation as....~ciated with the cholecystoduodenal fistula. All eight ~,atients recovered. The patient with gallstone ileus, :nanaged initially by only the removal of the obstruct:ag stone, subsequently underwent cholccystectomy ~nd closure of the duodenum. Of the eight patients Mth ¢holecystoduodenal fistula who underwent opera~ion, stones were removed from the common bile duct ~f one patient and operative cholangiography was reported as showing nothing abnormal in a second patient. In the six remaining patients the common bile duct was not opened at the time of operation. One of the six patients had an episode of chills, fever, and jaundice one month after operation, and postmortem examination of another patient who later died of aplastic anemia revealed calculi within the common bile duct. Operation was not performed in four patients with cholecystoduodenal fistula because of metastalic adenocarcinoma, terminal uremia, neuromuscular dystrophy, and multiple myeloma each in a single case. Three patients with cholecystoducxtenal fistula refused operation; two had severe cholangitis and the third had gallstone ileus. All three patients recovered; two t~f the three patients passed gallstones per rectum. One of the three patients was lost to follow-up study; ~hc other two have been asymptomatic for seven and ~'cn yeats, respectively. Operations were performed in all six patients with cholccystocolonic fistula. Four of the six underwent cholecystectomy and closure of the defect in the ;~cending colon. Cholecystectomy and right hemicolec~'~lny were performed in the patient with associated ;,olyps of the cecum. The patient who presented with :~cchanical obstruction of the ascending colon initially underwent cecostomy followed after one month v cholecystectomy and removal of a segment of the :~cending colon. Cholcdochotomy was performed in ~hrec of the six patients with cholecystocolonic fistula, ::nd calculi were found in the common bile duct in ..ach case. All six patients recovered after operation. The single patient with a choledochoduodenal fistula, seventy-five year old man with symptoms and signs
voL 120, July ]970
of recurrent pancreatitis, improved with medical management, was discharged from the hospital, and unfortunately was lost to follow-up study. Comments The relative incidence of each type of internal biliary fistula was demonstrated by Waggoner and Le Mone's review [I] of 819 eases in which they ftmnd 51 per cent were cholecystoduodenal, 21 per cent cholecysto=olonic, and 19 per cent choledochoduodenal. The remaining cases included cholecystogastric, choledochogastric, and cholecystocholedochal fistulas, in rare instances biliary fistulas have also been reported to extend into the renal pelvis, uterus, p~rtal vein, pericardium, and bronchial tree [2]. When all types of internal biliary fistulas are totaled, the over-all incidence is reported as 0.l to 0.5 per cent in autopsy series and 1.2 to 5.0 per cent in large series of operations to remove the gallbladder [2--4]. The fact that biliary-entefic listula will heal st~ntaneously may explain the lower incidence in autopsy studies. Some internal biliary fistulas are the result of operative or other trauma, but most occur spontaneously. Those fistulas which occur spontaneously between the gallbladder or bile ducts and the intestine are usually the result of chronic cholecystitis, peptic ulcer, or neoplasm. Since in most instances the etiology and problems in management of intestinal fistulas from the gallbladder differ from those of fistulas of the bile ducts, they should be considered and discussed as separate entities. The development of fistulous tracts from the gallbladder is associated with gallstones in as many as 90 per cent of cases [5]. Initially, adhesions between the gallbladder and the intestine are produced by recurrent attacks of cholecystitis. When inflammation and pressure of calculi cause necrosis of the wall of the gallbladder, the adherent portion of the intestine is destroyed in a similar manner and a fistula is formed. Because of its proximity, the duodenum is the most common portion of the intestine involved. As might be expected, the hepatic flexure of the colon is the next most frequent site of a fistula from disease of the gallbladder. Fistulas between the bile ducts and lhe duodenum, in contrast to those between the gallbladder and duodenum, are usually the result of peptic ulceration of the duodenum. As many as 80 per cent of choledochoduodenal fistulas are caused by penetrating duodenal ulcers which erode the anterior wall of the common bile duct [6]. Only in rare instances d o peplic ulcers cause a fistula between the duodenum and the gallbladder. In one series of thirty-six patients with biliary intestinal fistula secondary to peptic ulcer, thirty fistulas
29
Stull and Thomford were choledochoduodenal whereas only six were choleeystoduodenal [7]. There are no specific symptoms which suggest the development or presence of a biliary intestinal fistula. In some instances a fistula from the gallbladder into the duodenum may improve the condition of the patient by decompressing an obstructed and inflamed gallbladder. Symptoms, if any, from a choledochoduodenal fistula caused by peptic ulceration are usually overshadowed by the chronic recurrent symptoms of the ulcer. Exceptions to the common paucity of symptoms are the development of gallstone ileus, or, in a patient with cholecystocolonic fistula, the onset of diarrhea. Of the twenty-two patients described in this report, four had symptoms and signs of gallstone ileus. Only one of six patients with cholecystocolonic fistula had noted significant diarrhea. There are no studies of blood, serum, or urine which are diagnostic of biliary intestinal fistula, and identification of a fistula by cholecystography or cholangiography is rare. In contrast, roentgenograms of the abdomen both before and after a meal of barium are often diagnostic. In this series, air was seen in the biliary tree in 57 per cent of p~tients who had plain roentgenograms of the abdomen. Rocntgenograms after barium studies showed barium in the biliary tree in 69 per cent of the patients examined. When evaluating the individual case, however, it is important to recall that air in the biliary tree is not pathognomonic of a fistula [8]. In rare instances air in the bile ducts may be seen as a result of an incompetent sphincter of Oddi. Complications of biliary intestinal fistulas include gallstone ileus, ascending cholangitis, severe diarrhea in cases of cholecystocolenic fistula, and gastrointestinal hemorrhage [9,10]. Gallstone ileus is of particular concern since the mortality of this complication has been reported to be as great as 50 per cent in some series. Obstruction of the intestine by a gallstone, usually lodged in the terminal portion of the ileum, accounts for 1 to 5 per cent of all cases of intestinal obstructio~t. Most authors have advocated removal of the obstructing calculus at the initial operation with repair of the fistula at a later date. There have been exceptions to this rule [9]. The decision to repair the fistula in addition to relieving the intestinal obstruction should be an intraoperative one determined by the general condition of the patient, the characteristics of the inflammatory process surrounding the fistula, and the extent of the operation necessary to divide the fistulous tract, correct the underlying disease, and repair the involved organs. Although in most instances hemobilia is the result of rupture of an aneurysm of the cystic or hepatic
30
artery, a few cases of hemorrhage from biliary duodenal fistula have been reported [9,10]. Hemorrh,'tge from a cholecystocolonic fistula is more rare with only seven such cases having been reported [lO]. In at least one instance of cholecystocolonic fistula, severe hemorrhage occurred after erosion of the cystic artery. Operations for cholecystoduodenal and cholecystocolonic fistulas caused by inflammatory disease of the biliary tree are relatively standard when there are no related complications. Cholecystectomy should be performed and the colon or duodenum separated from the biliary tree and repaired. In selected cases, choleeystostomy rather than cholecystectomy may be necessary because of severe inflammation. In other instances, such as when a mass within the colon or gallbladder suggests that the fistula is the result of malignant disease, en bloc removal of the adjacent tissues may be appropriate. When biliary intestinal fistulas are caused by peptic ulceration, primary attention must be given to correction of the ulcer diathesis which caused the complication. When it is technically possible, hemigastrectomy, closure of the duodenum, vagotomy, and gastrojejunostomy are advisable. This provides acceptable management of the ulcer and excludes the area of the fistula from the gastrointestinal stream. Any additional procedures on the biliary tree or fistula at the initial operation will depend on the general condition of the patient and the intraoperative findings. Obstruction to the flow of bile must be alleviated if only by catheter drainage of the bile ducts. Ideally, the fistula should be divided and the duodenum repaired together with appropriate correction of disease or defects in the biliary tree. In some instances inflammation may have resulted in fibrosis with a permanent stricture of the common bile duct. These cases may require choledochoduodenostomy or choledochojcjunostomy. It is important to emphasize that in all cases of biliary intestinal fistula, the anatomy of the fistula and the presence or absence of calculi or other abnormalities within the biliary tree should be carefully assessed by intraoperative cholangiography.
Summary Biliary intestinal fistulas developed spontaneously in twenty-two patients; fifteen of the fistulas were cholecystoduodenal, six were cholecystocolonic, and one was choledochoduodenal. Fourteen of the twentytwo patients underwent an operation for the fistula or a related complication; there were no postoperative deaths. Fistulas from the gallbladder to the duodenum are most common and usually result from chronic cholecystitis. Fistulas of the common bile duct are in The American Journal Of Surgery
Biliary Intestinal Fistula most cases due to a peptic ulcer; with the exception L,f life-threatening complications, the primary goal in tt~cse cases is correction of the ulcer diatheses. References 1. Waggoner CM, LeMone DV: Clinical and roentgen aspects of internal biliary fistulas, Radiology 53: 31. 1949. 2. Puestow CB: Spontaneous internal biliary fistula. Ann Sur8 115: 1043. 1942. 3. Noskin EA, Strauss AA. Strauss SF: Spontaneous in. ternal biliary fistula: a review of the literature and report of two cases, Ann Surg 130: 270, ]949. 4. Pitman RG, Davies A: The clinical and radiological features of spontaneous internal biliary fistulae. Brit J Surg 50: 414, 1962-3.
V,~I, 120, July I970
5. HIcken N, Coray QB: Spontaneous gastrointestinal biltary fistulas. Surg Gynec Obstet 82: 723, ]946. 6. Hutchings VC, Wheeler JR. Puestow CB: Choledocho, duodenal fistula complicating duodenal ulcer. Arch Surg 73: 598, 1956. 7. Kourias GB, Choultaras A: Spontaneous gastrointestinal billary fistula complicating duodenal ulcer. Surg Gynec Obstet 119: 1013, 1964, 8. Constant E0 Turcotte JG: Choledochoduodenal fistula: the natural history and management of an unusual complication of peptic ulcer disease. Ann Surg 167: 220, 1968. 9. Carlson EG, Gates CY, Novacovich G; Spontaneous fis. tulas between the 8allbladder and gastrointestinal tract. Surg Gynec Obstet 101: 321, 1955. 10. Kaplan BJ: Massive lower 8astrointestinal hemorrhage from cholecystocolic fistula. Dis Colon Rectum 10: 191, 1967,
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