CASE REPORT
Silent Pancreatic Pseudocyst An Unusual Cause of Extrahepatic Biliary Obstruction James M. Falko, MD, Hagop S. Mekhjian, MD, FACP, and Fred B. Thomas, MD, FACP
Obstructive jaundice or
chronic
pancreatitis
c a u s e d by e i t h e r a c u t e is well
documented
(1, 2). F r e q u e n t l y it is d u e to e d e m a o r n a r r o w ing of the pancreatic portion of the common bile d u c t by t h e i n f l a m m a t o r y p r o c e s s (3). Occasionally, pancreatic pseudocysts may cause j a u n d i c e by o b s t r u c t i n g t h e c o m m o n bile duct. W h e n t h i s o c c u r s , it is n e a r l y a l w a y s a s s o c i a t e d with clinical and laboratory evidence of either a c u t e o r c h r o n i c p a n c r e a t i t i s . I n t h e l a r g e s t series o f 5 p a t i e n t s w i t h c o m m o n bile d u c t o b s t r u c t i o n d u e to p a n c r e a t i c p s e u d o c y s t , all h a d clinical e v i d e n c e o f p a n c r e a t i t i s . In fact, all h a d typical abdominal pain and x-ray evidence of pancreatic calcification(l). We describe a unique patient who presented with painless obs t r u c t i v e j a u n d i c e w i t h o u t clinical o r b i o c h e m i cal e v i d e n c e o f p a n c r e a t i t i s .
Laparotomy
re-
vealed that a small pancreatic pseudocyst was o b s t r u c t i n g t h e d i s t a l c o m m o n bile d u c t .
CASE REPORT A 33-year-old white woman was admitted to the Ohio State University Hospital with a 3-month history of painless jaundice, 40-1b weight loss, pruritis, and aeholic stools. The patient had been a moderate ethanol ingestor until 9 months before admission but emphatically denied any further alcohol intake since. She had no abdominal pain, nausea, vomiting, diarrhea, or exposure to hepatotoxins and had not received blood transfusions. She had been taking phenformin for 1 year for diabetes mellitus. Physical examination on admission showed a chronically ill, thin, and obFrom the Department of Medicine, The Ohio State University College of Medicine, Columbus, Ohio. Address for reprint requests: Dr. Fred B. Thomas, Division of Gastroenterology, N-937, University Hospitals, 410 West 10th Avenue, Columbus, Ohio 43210.
Digestive Diseases, Yol. 20, No. S (June 1975)
viously jaundiced patient whose weight was only 40 kg. Blood pressure was 130/70 mm Hg, pulse 78/rain, and oral temperature 37~ Pertinent physical findings included an enlarged firm liver with a total percussible dullness of 14 era. There was no abdominal tenderness or masses, and there were no peripheral stigmata of chronic liver disease. Stool guaiac was negative. The remainder of the examination was otherwise normal. Initial laboratory data showed a hemoglobin of 11.9 g/ 100 ml, leukocyte count of 7600/ram 3 and a reticulocyte count of 4%. Platelet count, prothrombin time, serum folate, and serum vitamin Btz were normal. Blood urea nitrogen was 12 rag/100 ml, creatinine 0.9 mg/100 ml, and creatinine clearance 105 ml/min. Fecal fat determination on a 3day high-fat diet showed a fecal fat excretion of 5 g/24 hr (normal < 6 g/24 br). A first-stage Schilling's test was normal, with 13% urinary excretion of 57Co-labeled vitamin Bt2 at 24 hours. Hepatitis-associated antigen (HAA) and a-fetoprotein determinations were negative. Total protein was 6.9 g/100 ml, with an albumin fraction of 2.8 g/100 ml. Urine urobilinogen was 15 mg/24 hr. Serum lactate dehydrogenase (LDH) was 319 units/mI (normal < 300 units/ ml), serum glutamic oxalacetic transaminase (SGOT) 176 Karmen units/ml (normal < 40 units/ml), and serum glutamic pyruvic transaminase (SGPT) 169 Karmen units/ ml (normal < 40 units/ml). Total serum bilirubin was 8.4 rag/100 ml, with a direct reacting fraction of 6.4 rag/100 ml. Serum amylase was 84 units/100 ml (normal< 100 units/100 ml), serum lipase 0.4 units/ml (normal < 1.5 units/ml), and 24-hour urinary amylase was 3200 units (normal < 3500 units). Amylase and lipase determinations on several subsequent occasions were likewise normal. Chest and plain abdominal x-ray studies were normal. There were no pancreatic calcifications. An upper-gastrointestinal x-ray examination showed slightly thickened mucosal folds of the duodenal C-loop (Figure 1). Barium enema was normal Technetium-99 liver-spleen scan was normal. Selenomethionine-75 pancreatic scan showed nonvisualization of the pancreas. Abdominal echogram was normal. Since the patient's obstructive jaundice did not recede over the ensuing 10 days and it was not known whether her jaundice was intra- or extrahepatic in origin, a transhepatic
583
FALKO ET AL
Fig 1. Upper-gastrointestinal x-ray film showing slightly thickened mucosal folds of the duodenal C-loop.
cholangiogram was performed. Marked compression of the distal common bile duct by a mass in the head of the pancreas was demonstrated (Figure 2). Exploratory laparotomy was performed on the same day, with a preoperative diagnosis of carcinoma of the head of the pancreas. At laparotomy, a pancreatic pseudocyst measuring approximately 3
584
cm in diameter and containing clear fluid was found to be obstructing the terminal portion of the common bile duct. There was no evidence of pancreatic carcinoma, and multiple lymph-nodes biopsies showed no evidence of tumor. A pancreatic biopsy showed fibrosis and focal calcification. The gallbladder was normal and there were no gallstones.
Digestive Diseases, Vol, 20, No. 6 (June 1975)
PANCREATIC PSEUDOCYST IN EXTRAHEPATIC BILIARY OBSTRUCTION
Fig 2. Transhepatic cholangiogram demonstrating compression of the common bile duct by a mass in the head of the pancreas.
A cholecystectomy and side-to-side choledochoduodenostomy were carried out. Postoperatively, the patient's iaundice cleared rapidly, liver size returned to normal, and after 10 days, tests of liver function were also normal.
Digestive Diseases, Vol. 20, No. 6 (June 1975)
DISCUSSION In m o s t reviews of p a n c r e a t i c pseudocyst, j a u n d i c e occurs in 8 - 1 7 % of the cases (4-7).
585
FALKO ET AL Table 1. Studied Cases of Common Bile Duct Obstruction Secondary to Pancreatic Pseudocyst*
Authors Gonzales et al (1)
Caravati et al (6) Mashib et al (8) Sidel et al (2) Weinstein et al (3)
Present report Total positive
Patient number 1 2 3 4 5 6 7 8 9 10 11 12 13
Recent ethanol history
Abdominal Abdominal pain mass + + + + +
+ + +
+ + + + + -9of13
+ + + + + ? -
+ + + ?
8of12
? ? ? ? 5of8
Pancreatic calcificationl+ + + + + + ? + ? + ? ? 8of9
Serum amylase Normal Normal Normal Increased Normal Normal Increased Normal Not d o n e Not d o n e Not d o n e Not d o n e Normal 2of9
Total serum bilirubin (mg/100ml) 8.1 1.8 10.7 15.8 11.1 15.6 4.6 8.6 3,8 16.6 6.1 5.6 8.4
* + represents finding present; - represents finding absent; ? represents data not available. j-Pancreatic calcification demonstrated by abdominal x-ray.
When present, jaundice is usually the result of either concomitant biliary tract obstruction from choledocholithiasis or associated hepatocellular disease--eg, alcoholic hepatitis. Only rarely, however, is the jaundice due to mechanical obstruction of the common bile duct by the pseudocyst p e r se. In this connection, approximately 12 cases of obstructive jaundice directly attributable to a pancreatic pseudocyst have been described (Table 1). It should be emphasized that all previously reported cases have been accompanied by clinical and biochemical evidence of pseudocyst formation or typical clinical features of pancreatitis, such as recent ethanol intake, abdominal pain or mass, pancreatic calcification, history of documented acute pancreatitis, and either hyperamylasemia or hyperamylasuria (1, 2, 6, 8). In the most thoroughly studied and best-documented report of 5 patients, Gonzalez et al suggest that obstructive jaundice in association with x~ray evidence of pancreatic calcification strongly favors the diagnosis of pancreatic pseudocyst (1). 586
Although our patient had not ingested alcohol in the 9 months preceding her admission to the hospital, it is likely the pseudocyst and histologically proven chronic pancreatitis were etiologically related to her prior ethanol abuse. Of interest, however, is the total lack of antecedent symptoms or signs suggesting acute pancreatitis or pseudocyst. There was no abdominal mass, and the results of repeated serum amylase, serum lipase, and urinary amylase determinations were within normal range. More importantly, this patient did not have abdominal pain. In this regard, previous studies documented asymptomatic or "painless pancreatitis" in approximately 5-10% of patients with proven chronic pancreatitis (9). However, the absence of radiologic evidence of pancreatic calcification, the lack of steatorrhea, the normal serum vitamin B12 level, and the normal Schilling's test also suggested that our patient did not have clinical and biochemical evidence of chronic pancreatitis or pancreatic insufficiency (10). The initial impression after transhepatic Digestive Diseases, Vol. 20, No. 6 (June 1975)
PANCREATIC PSEUDOCYST IN EXTRAHEPATIC BILIARY OBSTRUCTION
cholangiogram was that this patient had a carcinoma of the head of the pancreas. However, at laparotomy a relatively small pseudocyst was found to be obstructing the common bile duct. The present case therefore illustrates that (a) pancreatic pseudocysts may occur with silent or painless pancreatitis and that (b) pseudocysts may cause extrahepatic biliary obstruction in the absence of either clinical and laboratory evidence of pancreatitis or associated primary hepatobiliary disease. SUMMARY
Extrahepatic biliary obstruction due to mechanical obstruction of the common bile duct is a relatively rare complication of pancreatic pseudocyst. When jaundice does occur, clinical or laboratory evidence of associated primary hepatobiliary disease or acute pancreatitis has invariably been present. The patient described had a 3-month history of painless iaundice, 40-1b weight loss, pruritus, and hepatomegaly, but no clinical or biochemical evidence of acute or chronic pancreatitis. After initial evaluation, including an abdominal echogram and a transhepatic cholangiogram, carcinoma of the head of the pancreas was diagnosed preoperatively. At laparotomy, a small pancreatic pseudocyst obstructed the terminal portion of the common bile duct. This case illustrates that a pancreatic pseudocyst should be considered in the differential diagnosis of obstructive .jaundice, even in
Digestive Diseases, Vol. 20, No. 6 (June 1975)
the absence of clinical evidence of pancreatitis or pseudocyst formation. REFERENCES
1. Gonzalez LL, .Jaffe MS, Wiot,JF, Altemeier M: Pancreatic pseudocyst: A cause of obstructive jaundice. Ann Surg 161:569-576, 1965 2. Sidel VW, Wilson RE, Shipp JC: Pseudoeyst formation in chronic panereatitis. A cause of obstructive jaundice. Arch Surg 77:933-937, 1958 3. Weinstein BR, Korn RS, Zimmerman H J: Obstructive jaundice as a complication of pancreatitis. Ann Intern Med 58:245-258, 1963 4. Thomford N, Jesseph J: Pseudocyst of the pancreas: A review of 50 cases. Am J Surg 118:8693, 1969 5. Walker LG, Stone HH, Apple DG: Pseudocyst of the pancreas: A review of 59 cases. South Med J 60:389-393, 1967 6. Caravati CM, Ashworth JS, Frederick P: Pancreatic pseudocyst. A medical evaluation. JAMA 197:572-576, 1966 7. Rienhoff WF: An evaluation of pancreatic cysts treated at Johns Hopkins Hospital. Surgery 47:188-194, 1960 8. Mashib B, Lowenfels AB, Pendse PD, Rohman M: Jaundice from pancreatic pseudocyst. NY State J Med 71:2312-2313, 1971 9. Bartholamew LG, Comfort MW: Chronic panereatitis without pain. Gastroenterology 31:727745, 1956 10. Toskes PP, Hansel J, Cerda J, Deren J J: Vitamin B12 malabsorption in chronic pancreatic insufficiency. N Engl J Med 284:627-632, 1971
587
Silent Pancreatic Pseudocyst An Unusual Cause of Extrahepatic Biliary Obstruction James M. Falko, MD, Hagop S. Mekhjian, MD, FACP, and Fred B. Thomas, MD, FACP
Obstructive jaundice or
chronic
pancreatitis
c a u s e d by e i t h e r a c u t e is well
documented
(1, 2). F r e q u e n t l y it is d u e to e d e m a o r n a r r o w ing of the pancreatic portion of the common bile d u c t by t h e i n f l a m m a t o r y p r o c e s s (3). Occasionally, pancreatic pseudocysts may cause j a u n d i c e by o b s t r u c t i n g t h e c o m m o n bile duct. W h e n t h i s o c c u r s , it is n e a r l y a l w a y s a s s o c i a t e d with clinical and laboratory evidence of either a c u t e o r c h r o n i c p a n c r e a t i t i s . I n t h e l a r g e s t series o f 5 p a t i e n t s w i t h c o m m o n bile d u c t o b s t r u c t i o n d u e to p a n c r e a t i c p s e u d o c y s t , all h a d clinical e v i d e n c e o f p a n c r e a t i t i s . In fact, all h a d typical abdominal pain and x-ray evidence of pancreatic calcification(l). We describe a unique patient who presented with painless obs t r u c t i v e j a u n d i c e w i t h o u t clinical o r b i o c h e m i cal e v i d e n c e o f p a n c r e a t i t i s .
Laparotomy
re-
vealed that a small pancreatic pseudocyst was o b s t r u c t i n g t h e d i s t a l c o m m o n bile d u c t .
CASE REPORT A 33-year-old white woman was admitted to the Ohio State University Hospital with a 3-month history of painless jaundice, 40-1b weight loss, pruritis, and aeholic stools. The patient had been a moderate ethanol ingestor until 9 months before admission but emphatically denied any further alcohol intake since. She had no abdominal pain, nausea, vomiting, diarrhea, or exposure to hepatotoxins and had not received blood transfusions. She had been taking phenformin for 1 year for diabetes mellitus. Physical examination on admission showed a chronically ill, thin, and obFrom the Department of Medicine, The Ohio State University College of Medicine, Columbus, Ohio. Address for reprint requests: Dr. Fred B. Thomas, Division of Gastroenterology, N-937, University Hospitals, 410 West 10th Avenue, Columbus, Ohio 43210.
Digestive Diseases, Yol. 20, No. S (June 1975)
viously jaundiced patient whose weight was only 40 kg. Blood pressure was 130/70 mm Hg, pulse 78/rain, and oral temperature 37~ Pertinent physical findings included an enlarged firm liver with a total percussible dullness of 14 era. There was no abdominal tenderness or masses, and there were no peripheral stigmata of chronic liver disease. Stool guaiac was negative. The remainder of the examination was otherwise normal. Initial laboratory data showed a hemoglobin of 11.9 g/ 100 ml, leukocyte count of 7600/ram 3 and a reticulocyte count of 4%. Platelet count, prothrombin time, serum folate, and serum vitamin Btz were normal. Blood urea nitrogen was 12 rag/100 ml, creatinine 0.9 mg/100 ml, and creatinine clearance 105 ml/min. Fecal fat determination on a 3day high-fat diet showed a fecal fat excretion of 5 g/24 hr (normal < 6 g/24 br). A first-stage Schilling's test was normal, with 13% urinary excretion of 57Co-labeled vitamin Bt2 at 24 hours. Hepatitis-associated antigen (HAA) and a-fetoprotein determinations were negative. Total protein was 6.9 g/100 ml, with an albumin fraction of 2.8 g/100 ml. Urine urobilinogen was 15 mg/24 hr. Serum lactate dehydrogenase (LDH) was 319 units/mI (normal < 300 units/ ml), serum glutamic oxalacetic transaminase (SGOT) 176 Karmen units/ml (normal < 40 units/ml), and serum glutamic pyruvic transaminase (SGPT) 169 Karmen units/ ml (normal < 40 units/ml). Total serum bilirubin was 8.4 rag/100 ml, with a direct reacting fraction of 6.4 rag/100 ml. Serum amylase was 84 units/100 ml (normal< 100 units/100 ml), serum lipase 0.4 units/ml (normal < 1.5 units/ml), and 24-hour urinary amylase was 3200 units (normal < 3500 units). Amylase and lipase determinations on several subsequent occasions were likewise normal. Chest and plain abdominal x-ray studies were normal. There were no pancreatic calcifications. An upper-gastrointestinal x-ray examination showed slightly thickened mucosal folds of the duodenal C-loop (Figure 1). Barium enema was normal Technetium-99 liver-spleen scan was normal. Selenomethionine-75 pancreatic scan showed nonvisualization of the pancreas. Abdominal echogram was normal. Since the patient's obstructive jaundice did not recede over the ensuing 10 days and it was not known whether her jaundice was intra- or extrahepatic in origin, a transhepatic
583
FALKO ET AL
Fig 1. Upper-gastrointestinal x-ray film showing slightly thickened mucosal folds of the duodenal C-loop.
cholangiogram was performed. Marked compression of the distal common bile duct by a mass in the head of the pancreas was demonstrated (Figure 2). Exploratory laparotomy was performed on the same day, with a preoperative diagnosis of carcinoma of the head of the pancreas. At laparotomy, a pancreatic pseudocyst measuring approximately 3
584
cm in diameter and containing clear fluid was found to be obstructing the terminal portion of the common bile duct. There was no evidence of pancreatic carcinoma, and multiple lymph-nodes biopsies showed no evidence of tumor. A pancreatic biopsy showed fibrosis and focal calcification. The gallbladder was normal and there were no gallstones.
Digestive Diseases, Vol, 20, No. 6 (June 1975)
PANCREATIC PSEUDOCYST IN EXTRAHEPATIC BILIARY OBSTRUCTION
Fig 2. Transhepatic cholangiogram demonstrating compression of the common bile duct by a mass in the head of the pancreas.
A cholecystectomy and side-to-side choledochoduodenostomy were carried out. Postoperatively, the patient's iaundice cleared rapidly, liver size returned to normal, and after 10 days, tests of liver function were also normal.
Digestive Diseases, Vol. 20, No. 6 (June 1975)
DISCUSSION In m o s t reviews of p a n c r e a t i c pseudocyst, j a u n d i c e occurs in 8 - 1 7 % of the cases (4-7).
585
FALKO ET AL Table 1. Studied Cases of Common Bile Duct Obstruction Secondary to Pancreatic Pseudocyst*
Authors Gonzales et al (1)
Caravati et al (6) Mashib et al (8) Sidel et al (2) Weinstein et al (3)
Present report Total positive
Patient number 1 2 3 4 5 6 7 8 9 10 11 12 13
Recent ethanol history
Abdominal Abdominal pain mass + + + + +
+ + +
+ + + + + -9of13
+ + + + + ? -
+ + + ?
8of12
? ? ? ? 5of8
Pancreatic calcificationl+ + + + + + ? + ? + ? ? 8of9
Serum amylase Normal Normal Normal Increased Normal Normal Increased Normal Not d o n e Not d o n e Not d o n e Not d o n e Normal 2of9
Total serum bilirubin (mg/100ml) 8.1 1.8 10.7 15.8 11.1 15.6 4.6 8.6 3,8 16.6 6.1 5.6 8.4
* + represents finding present; - represents finding absent; ? represents data not available. j-Pancreatic calcification demonstrated by abdominal x-ray.
When present, jaundice is usually the result of either concomitant biliary tract obstruction from choledocholithiasis or associated hepatocellular disease--eg, alcoholic hepatitis. Only rarely, however, is the jaundice due to mechanical obstruction of the common bile duct by the pseudocyst p e r se. In this connection, approximately 12 cases of obstructive jaundice directly attributable to a pancreatic pseudocyst have been described (Table 1). It should be emphasized that all previously reported cases have been accompanied by clinical and biochemical evidence of pseudocyst formation or typical clinical features of pancreatitis, such as recent ethanol intake, abdominal pain or mass, pancreatic calcification, history of documented acute pancreatitis, and either hyperamylasemia or hyperamylasuria (1, 2, 6, 8). In the most thoroughly studied and best-documented report of 5 patients, Gonzalez et al suggest that obstructive jaundice in association with x~ray evidence of pancreatic calcification strongly favors the diagnosis of pancreatic pseudocyst (1). 586
Although our patient had not ingested alcohol in the 9 months preceding her admission to the hospital, it is likely the pseudocyst and histologically proven chronic pancreatitis were etiologically related to her prior ethanol abuse. Of interest, however, is the total lack of antecedent symptoms or signs suggesting acute pancreatitis or pseudocyst. There was no abdominal mass, and the results of repeated serum amylase, serum lipase, and urinary amylase determinations were within normal range. More importantly, this patient did not have abdominal pain. In this regard, previous studies documented asymptomatic or "painless pancreatitis" in approximately 5-10% of patients with proven chronic pancreatitis (9). However, the absence of radiologic evidence of pancreatic calcification, the lack of steatorrhea, the normal serum vitamin B12 level, and the normal Schilling's test also suggested that our patient did not have clinical and biochemical evidence of chronic pancreatitis or pancreatic insufficiency (10). The initial impression after transhepatic Digestive Diseases, Vol. 20, No. 6 (June 1975)
PANCREATIC PSEUDOCYST IN EXTRAHEPATIC BILIARY OBSTRUCTION
cholangiogram was that this patient had a carcinoma of the head of the pancreas. However, at laparotomy a relatively small pseudocyst was found to be obstructing the common bile duct. The present case therefore illustrates that (a) pancreatic pseudocysts may occur with silent or painless pancreatitis and that (b) pseudocysts may cause extrahepatic biliary obstruction in the absence of either clinical and laboratory evidence of pancreatitis or associated primary hepatobiliary disease. SUMMARY
Extrahepatic biliary obstruction due to mechanical obstruction of the common bile duct is a relatively rare complication of pancreatic pseudocyst. When jaundice does occur, clinical or laboratory evidence of associated primary hepatobiliary disease or acute pancreatitis has invariably been present. The patient described had a 3-month history of painless iaundice, 40-1b weight loss, pruritus, and hepatomegaly, but no clinical or biochemical evidence of acute or chronic pancreatitis. After initial evaluation, including an abdominal echogram and a transhepatic cholangiogram, carcinoma of the head of the pancreas was diagnosed preoperatively. At laparotomy, a small pancreatic pseudocyst obstructed the terminal portion of the common bile duct. This case illustrates that a pancreatic pseudocyst should be considered in the differential diagnosis of obstructive .jaundice, even in
Digestive Diseases, Vol. 20, No. 6 (June 1975)
the absence of clinical evidence of pancreatitis or pseudocyst formation. REFERENCES
1. Gonzalez LL, .Jaffe MS, Wiot,JF, Altemeier M: Pancreatic pseudocyst: A cause of obstructive jaundice. Ann Surg 161:569-576, 1965 2. Sidel VW, Wilson RE, Shipp JC: Pseudoeyst formation in chronic panereatitis. A cause of obstructive jaundice. Arch Surg 77:933-937, 1958 3. Weinstein BR, Korn RS, Zimmerman H J: Obstructive jaundice as a complication of pancreatitis. Ann Intern Med 58:245-258, 1963 4. Thomford N, Jesseph J: Pseudocyst of the pancreas: A review of 50 cases. Am J Surg 118:8693, 1969 5. Walker LG, Stone HH, Apple DG: Pseudocyst of the pancreas: A review of 59 cases. South Med J 60:389-393, 1967 6. Caravati CM, Ashworth JS, Frederick P: Pancreatic pseudocyst. A medical evaluation. JAMA 197:572-576, 1966 7. Rienhoff WF: An evaluation of pancreatic cysts treated at Johns Hopkins Hospital. Surgery 47:188-194, 1960 8. Mashib B, Lowenfels AB, Pendse PD, Rohman M: Jaundice from pancreatic pseudocyst. NY State J Med 71:2312-2313, 1971 9. Bartholamew LG, Comfort MW: Chronic panereatitis without pain. Gastroenterology 31:727745, 1956 10. Toskes PP, Hansel J, Cerda J, Deren J J: Vitamin B12 malabsorption in chronic pancreatic insufficiency. N Engl J Med 284:627-632, 1971
587
