CASE REPORT
Cholecystoduodenocolic Fistula Secondary to Carcinoma of the Gallbladder S T E V E N J. M O R R I S , MD, R I C H A R D A. G R E E N W A L D , MD, J A M I E S. B A R K I N , MD, F R A N C I S J. T E D E S C O , MD, and R O B E R T S N Y D E R , MD
C h o l e c y s t o d u o d e n o c o l i c fistulas are extremely unc o m m o n , with only 22 cases reported (1-6). Twenty-one of these fistulas have resulted from penetration by gallstones, and one was secondary to duodenal ulcer disease. This communication reports the 23rd case of this fistula and the first due to primary adenocarcinoma o f the gallbladder.
CASE REPORT A 55-year-old white man was admitted to the Miami Veterans Administration Hospital with complaints of diarrhea, weight loss, and weakness. One year prior to admission the patient developed intermittent right upper quadrant discomfort, and four months prior to admission he developed 8-10 black, watery, bowel movements per day. He lost 60 pounds over one year prior to admission despite persistance of a good appetite. He denied nausea, vomiting, fever, or history of gallbladder, liver, or ulcer disease. Physical examination revealed a cachectic, chronically ill appearing man in no acute distress. He was afebrile with a pulse of 80/rain, BP 150/80 mm Hg without orthostasis. The liver was 10 cm by palpation. There was a firm RUQ mass which could not be felt separately from the liver edge. Stool was guaiac positive. Laboratory data included: hemoglobin 3.8 g/100 ml, hematocrit 16 volume %, and leukocytes 9800/mm 3. Peripheral smear disclosed hypochromic, microcytic cells. Electrolytes, glucose, BUN, prothrombin time, and partial thromboplastin time were normal. The albumin From the University of Miami School of Medicine and Veterans Administration Hospital, Department of Medicine, Division of Gastroenterology and Department of Surgery. Address for reprint requests: Dr. Jamie S. Barkin, Assistant Professor of Medicine, Division of Gastroenterology, University of Miami School of Medicine, Post Office Box 520875, Miami, Florida 33152.
was 2.6 g/100 ml, cholesterol 94 mg/100 ml, bilirubin 0.4 mg/100 ml, alkaline phosphatase 121 mU/ml, SGOT 18 mU/ml, and amylase 40 Somogyi units. Hospital Course. A barium enema examination revealed a fistula between the area of the hepatic flexure and the duodenum with no colonic mass demonstrated. The fistula on the upper-gastrointestinal tract was seen to enter the second part of the duodenum (Figure 1). Colonoscopy was performed and several fistulous tracts were visualized in the transverse colon, but the surrounding colonic mucosa appeared normal. Upper-gastrointestinal endoscopy revealed a submucosal mass in the second portion of the duodenum with a central fistula. Biopsies of this area showed inflammatory changes. Laparotomy disclosed a mass in the right upper quadrant with fistulous connections between a noncalculous gallbladder, duodenum, and colon (Figure 2). Sections of the mass showed mucinous adenocarcinoma of the gallbladder with invasion and fistulization to the duodenum and colon. Surgery included radical excision of the gallbladder bed, cholecystectomy, partial gastrectomy, vagotomy, duodenectomy, proximal pancreatectomy, right hemicolectomy, and resection of the proximal 8 in. of the jejunum. An anticolic antiperistaltic gastrojejunostomy (Polya), end-to-side choledochojejunostomy, and an ileotransverse colostomy were performed. Four months postoperatively the patient continues to gain weight and feel well.
DISCUSSION The most c o m m o n biliary-enteric fistulas are cholecystoduodenal, 60% of cases; cholecystocolic, 20%; and cholecystogastric, 10% (1). Multiple fistulas are distinctly unusual and are of two types. Patients may have c o n c o m m i t a n t fistula, ie, a cholecystoduodenal and cholecystocolic, but without
Digestive Diseases, Vol. 23, No. 9 (September 1978)
0002-9211/78/0900-0849505.00/19 1978DigestiveDisease Systems, Inc.
849
MORRIS ET AL
Fig 1. Upper-gastrointestinal series which reveals a fistula between the duodenum and hepatic flexure. (S, stomach; D, duodenum; F, fistula; C, colon).
communication between the duodenum and colon (7). Secondly, multiple fistula may exist joining more than two viscera in an abnormal connection, as in our patient. Gallstones eroding into the adjacent viscera are responsible for 75-90% of spontaneous biliary-enteric fistula (5). Primary carcinoma of the gallblad850
der is responsible for less than 5% of biliary-enteric fistula. Primary carcinoma of the gallbladder is, however, complicated by fistulization in a 3.5-4% of patients (8). The symptoms of carcinoma of the gallbladder are vague and nonspecific. Correct diagnosis prior to surgery or postmortem is reached only 5-10% of Digestive Diseases, Vol. 23, No. 9 (September 1978)
CHOLECYSTODUODENOCOLIC FISTULA
Fig 2. Pathology specimen showing opened gallbladder with gelatinous carcinoma. Fistula to the colon is evident (arrows).
the time. The tumor spreads both by lymphatics and direct extension into adjacent viscera with the liver involved in over 60% of cases. The gallbladder, in 80% of normal individuals, anatomically lies close to the superior medial aspect of the right colic flexure and to the second portion of the duodenum. Infiltration of the superior aspect of the hepatic flexure primarily occurs when a carcinoma originates in Digestive Diseases, Vol. 23, No. 9 (September 1978)
or involves the inferior surface of the body and fundus of the gallbladder. A carcinoma developing in the neck of the gallbladder has a greater tendency to extend into the duodenum (9). The major clinical presentation of cholecystoduodenocolic fistula is diarrhea and weight loss. The diarrhea has been attributed to a number of causes, including: (1) bile entering directly into the colon
851
MORRIS ET AL
causing a colitis (10); (2) direct loss of gastric contents into the colon; (3) fecal material refluxing into the small bowel resulting in jejunitis; and (4) loss of the bile pool with resultant steatorrhea (11). Another prominent symptom, not noted in our patient, is vomiting of feculent material or foul eructation. In addition, a major presentation of cholecystoduodenal fistula is small-bowel obstruction due to gallstone ileus. However, this has only been reported in four cases of cholecystoduodenocolic fistula (6). The diagnosis of biliary-enteric fistulas is primarily based on radiologic studies. Pneumobilia is a key finding with biliary-enteric fistula, apparent in up to 75% of cases. Barium enema examination will disclose the duodenocolic fistula in up to 90% of cases (12). Upper-gastrointestinal series, by contrast, may fail to disclose 50% of the fistulas. An oral cholecystogram, omitted in this case, is strongly urged in any patient with a duodenocolic fistula as failure to visualize the gallbladder makes involvement of this organ a serious consideration. Optimal therapy of carcinoma of the gallbladder is curative resection which, unfortunately, by the time of diagnosis is possible in only 15-30% of the tumors. This accounts for a 5-year survival of 7% (8). In patients with a cholecystoduodenocolic fistula from benign disease, surgical treatment consists of transection of the fistulas to the colon and duodenum, cholecystectomy, and careful search and removal of stones, if present, in the small bow, el. In summary we have presented a case of chole-
852
cystoduodenocolic fistula caused by adenocarcinoma of the gallbladder. The presentation was diarrhea and weight loss with a past history of right upper quadrant pain. Barium enema disclosed the duodenocolic communication initially.
REFERENCES 1. Dormal NM, Estacio R, Sherman H: Cholecystoduodenocolic fistula with gallstone ileus. Dis Colon Rectum 18:702704, 1975 2. Everingham S: Multiple internal biliary fistula. Surg Clin North Am 7:1349-1350, 1927 3. Pitman RG, Davies A: The Clinical and radiological features of spontaneous internal biliary fistulae. Br J Surg 508:414425, 1963 4. Amoury RA, Barker HG: Multiple biliary enteric fistulas. Am J Surg 111:180-185, 1966 5. Nemhauser GM, Thompson JC: Cholecystoduodenocolic fistula due to gallstones. Ann Surg 163:81-85, 1966 6. Schocket E, Evans J, Jonas S: Cholecystoduodenocolic fistula with gallstone ileus. Arch Surg 101:523-526, 1970 7. Burson LC, Berliner SD: Multiple simultaneous cholecystoenteric fistulas. NY State J Med 76:955-977, 1976 8. Vaittinen E: Carcinoma of the gallbladder. A study of 390 cases diagnosed in Finland 1953-1967. Ann Chit Gynaecol Fenn 168(Suppl 1):1-28, 1970 9. Gharemani G, Meyers MA: The cholecystocolic relationships. Am J Roentgenol Radium Ther Nuc Med 125(1):2134, 1975 10. Grayson MJ, O'Connel ND: Benign duodenocolic fistulae. Postgrad Med 36:549-557, 1960 11. Waggoner CM, LeMore DV: Clinical and roentgen aspects of internal biliary fistulas. Radiology 53:31-41, 1949 12. Brindle MJ, Kane JF: Benign duodenocolic fistula. Br J Surg 53:749-753, 1966
Digestive Diseases, Vol. 23, No. 9 (September 1978)
Cholecystoduodenocolic Fistula Secondary to Carcinoma of the Gallbladder S T E V E N J. M O R R I S , MD, R I C H A R D A. G R E E N W A L D , MD, J A M I E S. B A R K I N , MD, F R A N C I S J. T E D E S C O , MD, and R O B E R T S N Y D E R , MD
C h o l e c y s t o d u o d e n o c o l i c fistulas are extremely unc o m m o n , with only 22 cases reported (1-6). Twenty-one of these fistulas have resulted from penetration by gallstones, and one was secondary to duodenal ulcer disease. This communication reports the 23rd case of this fistula and the first due to primary adenocarcinoma o f the gallbladder.
CASE REPORT A 55-year-old white man was admitted to the Miami Veterans Administration Hospital with complaints of diarrhea, weight loss, and weakness. One year prior to admission the patient developed intermittent right upper quadrant discomfort, and four months prior to admission he developed 8-10 black, watery, bowel movements per day. He lost 60 pounds over one year prior to admission despite persistance of a good appetite. He denied nausea, vomiting, fever, or history of gallbladder, liver, or ulcer disease. Physical examination revealed a cachectic, chronically ill appearing man in no acute distress. He was afebrile with a pulse of 80/rain, BP 150/80 mm Hg without orthostasis. The liver was 10 cm by palpation. There was a firm RUQ mass which could not be felt separately from the liver edge. Stool was guaiac positive. Laboratory data included: hemoglobin 3.8 g/100 ml, hematocrit 16 volume %, and leukocytes 9800/mm 3. Peripheral smear disclosed hypochromic, microcytic cells. Electrolytes, glucose, BUN, prothrombin time, and partial thromboplastin time were normal. The albumin From the University of Miami School of Medicine and Veterans Administration Hospital, Department of Medicine, Division of Gastroenterology and Department of Surgery. Address for reprint requests: Dr. Jamie S. Barkin, Assistant Professor of Medicine, Division of Gastroenterology, University of Miami School of Medicine, Post Office Box 520875, Miami, Florida 33152.
was 2.6 g/100 ml, cholesterol 94 mg/100 ml, bilirubin 0.4 mg/100 ml, alkaline phosphatase 121 mU/ml, SGOT 18 mU/ml, and amylase 40 Somogyi units. Hospital Course. A barium enema examination revealed a fistula between the area of the hepatic flexure and the duodenum with no colonic mass demonstrated. The fistula on the upper-gastrointestinal tract was seen to enter the second part of the duodenum (Figure 1). Colonoscopy was performed and several fistulous tracts were visualized in the transverse colon, but the surrounding colonic mucosa appeared normal. Upper-gastrointestinal endoscopy revealed a submucosal mass in the second portion of the duodenum with a central fistula. Biopsies of this area showed inflammatory changes. Laparotomy disclosed a mass in the right upper quadrant with fistulous connections between a noncalculous gallbladder, duodenum, and colon (Figure 2). Sections of the mass showed mucinous adenocarcinoma of the gallbladder with invasion and fistulization to the duodenum and colon. Surgery included radical excision of the gallbladder bed, cholecystectomy, partial gastrectomy, vagotomy, duodenectomy, proximal pancreatectomy, right hemicolectomy, and resection of the proximal 8 in. of the jejunum. An anticolic antiperistaltic gastrojejunostomy (Polya), end-to-side choledochojejunostomy, and an ileotransverse colostomy were performed. Four months postoperatively the patient continues to gain weight and feel well.
DISCUSSION The most c o m m o n biliary-enteric fistulas are cholecystoduodenal, 60% of cases; cholecystocolic, 20%; and cholecystogastric, 10% (1). Multiple fistulas are distinctly unusual and are of two types. Patients may have c o n c o m m i t a n t fistula, ie, a cholecystoduodenal and cholecystocolic, but without
Digestive Diseases, Vol. 23, No. 9 (September 1978)
0002-9211/78/0900-0849505.00/19 1978DigestiveDisease Systems, Inc.
849
MORRIS ET AL
Fig 1. Upper-gastrointestinal series which reveals a fistula between the duodenum and hepatic flexure. (S, stomach; D, duodenum; F, fistula; C, colon).
communication between the duodenum and colon (7). Secondly, multiple fistula may exist joining more than two viscera in an abnormal connection, as in our patient. Gallstones eroding into the adjacent viscera are responsible for 75-90% of spontaneous biliary-enteric fistula (5). Primary carcinoma of the gallblad850
der is responsible for less than 5% of biliary-enteric fistula. Primary carcinoma of the gallbladder is, however, complicated by fistulization in a 3.5-4% of patients (8). The symptoms of carcinoma of the gallbladder are vague and nonspecific. Correct diagnosis prior to surgery or postmortem is reached only 5-10% of Digestive Diseases, Vol. 23, No. 9 (September 1978)
CHOLECYSTODUODENOCOLIC FISTULA
Fig 2. Pathology specimen showing opened gallbladder with gelatinous carcinoma. Fistula to the colon is evident (arrows).
the time. The tumor spreads both by lymphatics and direct extension into adjacent viscera with the liver involved in over 60% of cases. The gallbladder, in 80% of normal individuals, anatomically lies close to the superior medial aspect of the right colic flexure and to the second portion of the duodenum. Infiltration of the superior aspect of the hepatic flexure primarily occurs when a carcinoma originates in Digestive Diseases, Vol. 23, No. 9 (September 1978)
or involves the inferior surface of the body and fundus of the gallbladder. A carcinoma developing in the neck of the gallbladder has a greater tendency to extend into the duodenum (9). The major clinical presentation of cholecystoduodenocolic fistula is diarrhea and weight loss. The diarrhea has been attributed to a number of causes, including: (1) bile entering directly into the colon
851
MORRIS ET AL
causing a colitis (10); (2) direct loss of gastric contents into the colon; (3) fecal material refluxing into the small bowel resulting in jejunitis; and (4) loss of the bile pool with resultant steatorrhea (11). Another prominent symptom, not noted in our patient, is vomiting of feculent material or foul eructation. In addition, a major presentation of cholecystoduodenal fistula is small-bowel obstruction due to gallstone ileus. However, this has only been reported in four cases of cholecystoduodenocolic fistula (6). The diagnosis of biliary-enteric fistulas is primarily based on radiologic studies. Pneumobilia is a key finding with biliary-enteric fistula, apparent in up to 75% of cases. Barium enema examination will disclose the duodenocolic fistula in up to 90% of cases (12). Upper-gastrointestinal series, by contrast, may fail to disclose 50% of the fistulas. An oral cholecystogram, omitted in this case, is strongly urged in any patient with a duodenocolic fistula as failure to visualize the gallbladder makes involvement of this organ a serious consideration. Optimal therapy of carcinoma of the gallbladder is curative resection which, unfortunately, by the time of diagnosis is possible in only 15-30% of the tumors. This accounts for a 5-year survival of 7% (8). In patients with a cholecystoduodenocolic fistula from benign disease, surgical treatment consists of transection of the fistulas to the colon and duodenum, cholecystectomy, and careful search and removal of stones, if present, in the small bow, el. In summary we have presented a case of chole-
852
cystoduodenocolic fistula caused by adenocarcinoma of the gallbladder. The presentation was diarrhea and weight loss with a past history of right upper quadrant pain. Barium enema disclosed the duodenocolic communication initially.
REFERENCES 1. Dormal NM, Estacio R, Sherman H: Cholecystoduodenocolic fistula with gallstone ileus. Dis Colon Rectum 18:702704, 1975 2. Everingham S: Multiple internal biliary fistula. Surg Clin North Am 7:1349-1350, 1927 3. Pitman RG, Davies A: The Clinical and radiological features of spontaneous internal biliary fistulae. Br J Surg 508:414425, 1963 4. Amoury RA, Barker HG: Multiple biliary enteric fistulas. Am J Surg 111:180-185, 1966 5. Nemhauser GM, Thompson JC: Cholecystoduodenocolic fistula due to gallstones. Ann Surg 163:81-85, 1966 6. Schocket E, Evans J, Jonas S: Cholecystoduodenocolic fistula with gallstone ileus. Arch Surg 101:523-526, 1970 7. Burson LC, Berliner SD: Multiple simultaneous cholecystoenteric fistulas. NY State J Med 76:955-977, 1976 8. Vaittinen E: Carcinoma of the gallbladder. A study of 390 cases diagnosed in Finland 1953-1967. Ann Chit Gynaecol Fenn 168(Suppl 1):1-28, 1970 9. Gharemani G, Meyers MA: The cholecystocolic relationships. Am J Roentgenol Radium Ther Nuc Med 125(1):2134, 1975 10. Grayson MJ, O'Connel ND: Benign duodenocolic fistulae. Postgrad Med 36:549-557, 1960 11. Waggoner CM, LeMore DV: Clinical and roentgen aspects of internal biliary fistulas. Radiology 53:31-41, 1949 12. Brindle MJ, Kane JF: Benign duodenocolic fistula. Br J Surg 53:749-753, 1966
Digestive Diseases, Vol. 23, No. 9 (September 1978)
