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PubMed2002

Choledochal cysts in adults and the importance of differential diagnosis.

BACKGROUND/PURPOSE: Choledochal cysts, congenital cystic dilatations of the hepatobiliary tree, present diagnostic dilemmas. In countries where hydatid disease is endemic, this rare pathology may be overlooked. The importance of this issue led us to reexamine our clinical material to evaluate our findings and management. METHODS: This study examined 12 patients (11 females, 1 male) who were treated at the University of Istanbul, Cerrahpasa Medical Faculty, with the diagnosis of choledochal cyst between 1981 and 2000. The cases were examined retrospectively, and the complaints, diagnostic methods, findings, and management were discussed. RESULTS: Ten of the cysts were type I (83.3%), one was type II (8.3%), and one was type IV (8.3%). Three of the patients underwent operation with the diagnosis of liver hydatid cyst, but during the operation the cysts were found to be choledochal cysts. In six of the patients, the cysts were correctly diagnosed, and they were excised. CONCLUSIONS: Contemporary approaches to treating choledochal cysts focus on total removal of the cyst. In a country where hydatic diseases are endemic, hepatobiliary cystic lesions are often misdiagnosed clinically and radiologically as hydatid cysts. Considering that percutaneous and laparoscopic approaches are being increasingly adopted for treatment of hydatid cysts, the problematic area must be subjected to thorough examination for cystic lesions. The possibility of choledochal cysts must always be kept in mind; otherwise, an unfavorable process may begin for both patient and surgeon.

Adolescent↗
PubMed1989

Etiological relationships between choledochal cyst and anomalous junction of the pancreaticobiliary ductal system.

The etiology of choledochal cyst has been investigated by clinicians and pathologists primarily of Germany, Austria, United States, United Kingdom, and Japan from the end of the last century, but Japanese investigators contributed greatly to the study of the relationship between choledochal cyst and anomalous junction of the pancreaticobiliary ductal system. Heid (1893) is considered to be the first to document choledochal cyst associated with anomalous pancreaticobiliary junction, but it was probably Kozumi et al. (1916) that first evaluated anomalous junction in relation to the etiology of choledochal cyst. Yotsuyanagi (1936), who is noted for his work on the etiological studies of choledochal cyst, attached particular importance to anomalous junction and attempted to establish a relationship between dysplasia of the primordial common bile duct and its anomalous junction with the pancreatic duct. It is no exaggeration to say that this area was led by Japanese investigators even after the introduction of Babbitt's anomalous junction theory (1968). In this review, etiological theories of choledochal cyst and their changes with time were discussed separately for those before and after the advent of Babbitt's anomalous junction theory (1968) with particular reference to the causative relationship between anomalous junction and choledochal cyst. Although there is as yet no definite answer to this problem, the theories advanced to date are considered to be summarized as the following three major lines of thought: 1) bile duct dilatation and anomalous junction are both congenital dysplasia occurring simultaneously during the embryonic development, 2) bile duct dilatation is a secondary change induced by influx of pancreatic juice into the biliary tract due to anomalous pancreaticobiliary junction, and 3) bile duct dilatation is induced secondarily by stenosis of the distal bile duct, which is a part of complex anomaly of the pancreaticobility system. Which of these etiological mechanisms is more important than others must still be clarified, but these three factors are considered to be interrelated with one another in the etiology of choledochal cyst.

Biliary Fistula↗
PubMed2005

Single-stage reconstruction of perforated choledochal cyst: case report and review of the literature.

Choledochal cysts represent a rare disease in the Western world. We reviewed our recent experience with a case of perforated choledochal cyst, define the currently accepted treatment options, and review the literature of this unusual disease. An 11-month-old girl presented with abdominal pain and distention as well as non-bilious vomiting. Subsequent workup included endoscopic retrograde cholangiopancreatography revealing a perforated type I choledochal cyst. She underwent single-stage excision and reconstruction with a Roux-en-Y hepaticojejunostomy. Perforated choledochal cyst is a rare event, and prompt surgical intervention is warranted. Single-stage cystectomy and Roux-en-Y reconstruction is possible in select patients. A thorough understanding of the pathophysiology, management, and follow-up is required.

Biliary Tract Surgical Procedures↗
PubMed2005

Type IV-A choledochal cysts: a challenge.

BACKGROUND/PURPOSE: Type IV-A choledochal cysts are characterized by congenital cystic dilatation of the biliary tree extending to involve the intrahepatic biliary channels also. A single-center experience of the management of type IV-A choledochal cysts is presented. METHODS: Thirty-five out of 105 (33%) patients with choledochal cysts, who underwent surgery at a tertiary care center in northern India from January 1989 to December 2002, were found to have a type IV-A (Todani's classification) cyst. The mean age of the patients was 24 years (range, 3 months to 60 years); 17 patients in the group were adults and 22 were females. Presenting features were abdominal pain, jaundice, cholangitis, and abdominal lump, in various combinations. RESULTS: Excision of the extrahepatic part of the cyst and a wide bilio-enteric anastomosis was achieved in 32 (91%) patients, while internal drainage of the cyst was necessitated in 3 patients, for technical reasons: collaterals due to portal hypertension (1 patient) and dense adhesions (2 patients). Six (17%) patients developed postoperative complications: 3 had bilio-enteric anastomosis leaks, with 2 requiring a percutaneous proximal biliary diversion; 2 had intraabdominal bleeds requiring re-exploration, and 1 had external pancreatic fistula that closed spontaneously. Follow-up information was available for 28 (80%) patients. Mean duration of follow up was 25 months (median, 12 months; range, 6 months to 9 years). Three patients required re-operation, for anastomotic stricture (n = 2) and hepatolithiasis and recurrent cholangitis (n = 1) during follow up. CONCLUSIONS: Excision of the extrahepatic part of the cyst and drainage of the intrahepatic part by a wide hilar or subhilar anastomosis gave satisfactory results in the majority of patients with type IV-A choledochal cysts. Close long-term follow up of these patients is essential, because they are likely to present with complications related to the residual intrahepatic part of the disease.

Adolescent↗
PubMed2000

[Choledochal cysts: surgical treatment].

The excision of the choledochal cyst with bile drainage through intestinal conduit is a standard operative procedure in the surgical management of choledochal cysts. During the last eight years five patients have been treated with this operation at the University Children's Hospital in Zagreb. All the patients were girls aged from two months to twelve years. The classical triad of pain, jaundice and abdominal mass was observed in only one patient, an eight-year-old girl. The only symptom in infancy was jaundice. Diagnosis was made by abdominal ultrasound, bibliography, CT scan with hepatotropic contrast and in older children by ERCP. Four cysts were type Todani I, and one cyst was Todani type II. The complete excision of the choledochal cyst with the Roux-Y jejunal conduit without antireflux valve was performed. There was neither operative morbidity nor mortality. Three months postoperatively the control ultrasonography and liver laboratory tests were without abnormalities. The routine control which followed did not show episodes of cholangitis, lithiasis, lipid malabsorption, blood clotting abnormalities or growth failure. The complete excision of the cyst with Roux-Y hepaticoenterostomy is an operative treatment with good results in infancy and childhood.

Child↗
PubMed1996

Early prenatal diagnosis of choledochal cyst.

Unrecognized biliary tract obstruction due to choledochal cysts or biliary atresia can result in significant morbidity and mortality. Diagnosis in utero of choledochal cyst allows prompt postnatal diagnostic evaluation and appropriate surgical therapy to be instituted early in life. This may minimize the potential complications of cholangitis, cirrhosis, and liver failure in infants with choledochal cysts.

Adult↗
PubMed1979

Portal hypertension secondary to choledochal cyst.

Two patients with portal hypertension secondary to choledochal cyst are reported and combined with four others previously reported in the literature. Choledochal cysts may, by extrensic compression, cause partial or complete portal vein obstruction, portal hypertension and esophageal varices. Needle biopsy of the liver is probably not a reliable means of differentiating choledochal cyst from other intrahepatic causes of portal hypertension. Internal drainage of the choledochal cyst has been performed in four patients and in each instance resulted in satisfactory portal decompression.

Adolescent↗
PubMed1994

A case of choledochal cyst associated with adenocarcinoma exhibiting sarcomatous features.

One complication of choledochal cyst in adulthood is the development of secondary carcinoma, usually well-differentiated adenocarcinoma. We report a case of extrahepatic bile duct carcinoma associated with choledochal cyst and presenting predominantly sarcomatous features. The patient was a 52-year-old female who presented with abdominal fullness and pain. Ultrasound (US), computed tomography (CT), and cholangiography revealed a choledochal cyst with a tumor. The resected choledochal cyst was 10 x 7.5 cm and contained a protruding, ulcerated tumor, measuring about 4.0 x 2.5 cm. Histologically, the tumor was composed of spindle-shaped or fusiform cells with occasional pleomorphic or bizarre giant cells and abundant fibrous stroma, similar to malignant mesenchymal tumors. However, further histologic and immunohistochemical examination revealed that the tumor was adenocarcinoma with prominent sarcomatous features. To our knowledge, sarcomatous change in extrahepatic bile duct carcinoma associated with choledochal cyst has not been previously described.

Adenocarcinoma↗
PubMed1997

Choledochal cyst and neoplasm: a comprehensive review of 106 cases and presentation of two original cases.

Choledochal cysts are prone to complications: cholangitis, biliary cirrhosis, portal hypertension, lithiasis, rupture, pancreatitis, and carcinoma. The coincidence of choledochal cysts and neoplasia ranges from 2.5 to 26 per cent. One hundred six cases of choledochal cysts with neoplasms have been collected from the literature. We have tabulated the results of 68 cases found to have a neoplasm at the initial laparotomy and of 38 patients with pristine choledochal cysts treated electively who subsequently developed a neoplasm. Two original cases are presented. The primary site of neoplasia was not confined only to choledochal cysts. There appears to be a propensity for malignancy to develop anywhere in the biliary tract or gallbladder or pancreas in conjunction with the choledochal cyst. Accompanying choledochal cysts is a high incidence of an anomalous relation at the pancreaticobiliary junction with subsequent malignancy formation. A pathogenetic basis is postulated.

Adenocarcinoma↗
PubMed1997

Reoperative surgery for choledochal cysts.

BACKGROUND: Complications often follow if a choledochal cyst is treated simply by drainage, either internal or external. This study reviews 17 patients who had had previous cystoenterostomy (n = 9) or external drainage (n = 8) and who required reoperation and cyst excision. METHODS: The study was a retrospective review including ten women and seven men managed over 9 years. The indications for reoperation were stone formation (10 patients), pancreatitis (three), portal hypertension (two) and hepatic abscess (one); two patients were asymptomatic. RESULTS: Definitive surgery with cyst excision was possible in all patients who had previously had external cyst drainage and in seven of nine who had had previous cystoenterostomy. There were no deaths. Two postoperative biliary leaks and two duodenal fistulas resolved spontaneously. CONCLUSION: Excision of a choledochal cyst is possible and desirable even after a previous drainage operation. In severely ill patients with a complication of choledochal cyst, external drainage may be a preferable initial manoeuvre.

Adolescent↗
PubMed2003

Abnormal pancreatic isoamylases in the serum of children with choledochal cyst.

PURPOSE: To investigate the changes of pancreatic isoamylases in the serum of children with choledochal cyst. METHODS: Serum and bile from 20 children with choledochal cyst were studied for total amylase activity and isoamylases. Using electrophoresis, the assay of pancreatic isoamylases was expressed by peak appearance rate (PAR) and ratio of isoenzyme activity (RIA). Serum from 20 healthy children and 12 adults with acute pancreatitis served as a comparison. RESULTS: The total amylase activity in the serum of children with choledochal cyst was slightly higher than that of healthy controls, but dramatically lower than that of acute pancreatitis. The serum of children with choledochal cyst showed 5 pancreatic peaks (PAR: P1, 100 %; P2, 100 %; P3, 100 %; P4, 75 %; P5, 45 %) without any major peak. In healthy controls, only P1 and P2 were present (PAR: P1, 40 %; P2, 100 %) with the latter being a major peak (RIA: 81.8 +/- 25.3 %). These results in children with choledochal cyst differed from those of patients with acute pancreatitis who presented with the less common P4 and no P5 (PAR: P1, 100 %; P2, 100 %; P3, 91.6 %; P4, 8.3 %). The pattern of pancreatic isoenzymes in bile was similar to that in the serum from patients with choledochal cyst. These abnormal pancreatic isoamylases gradually disappeared after operation. CONCLUSIONS: Abnormal pancreatic isoamylases, especially P4 and P5, are found in the serum of patients with choledochal cyst, which would point to a reflux of pancreatic juice into the biliary tract.

Abdominal Pain↗
PubMed1997

Manifestations and surgical treatment of choledochal cyst in different age group patients.

Whether early surgery is necessary for asymptomatic choledochal cyst is still controversial. In order to evaluate the manifestations of choledochal cyst on different age group patients, we investigated 57 patients, 36 females and 21 males, who underwent surgery for choledochal cyst at National Taiwan University Hospital from July 1988 to June 1995. Of these, 93% were classified as type I according to the Alonzo-Lej or Todani classification method. Patients were divided into three groups according to their age at surgery: group I, 14 infants (< 1-yr); group II, 28 children (1-16 yr); and group III, 15 adults (> 16 yr). The most common symptom in infants was jaundice, while abdominal pain was the most frequent in children and adults. Preoperative evaluation of liver function showed that alanine aminotransferase was higher in group II (160 +/- 77 IU/L) and group III (164 +/- 75 IU/L) than group I (74 +/- 28 IU/L). A higher tendency of biliary sludge and stone formation was noticed in group III (6/15, 40%) when compared with group I (2/14, 14.3%) and group II (5/28, 17.9%). The incidence of high bile amylase concentration in the choledochal cyst was higher in groups II (16/21,76.2%) and III (9/11, 81.8%) than in group I (2/9, 22.2%). There was more inflammatory changes detected in the choledochal cyst wall and gall bladder by histologic examination in older patients (groups II and III). Therefore, an early surgery for choledochal cyst is suggested for prevention of liver injury, biliary sludge, stone formation and pancreatitis.

Adolescent↗
PubMed1998

Type I choledochal cyst in association with familial adenomatous polyposis.

Choledochal cysts and familial adenomatous polyposis are infrequent disorders that are often manifest in childhood or in early adult life. The rarity and early presentation of these diseases suggests a genetic basis, which has been established for familial polyposis but not for choledochal cysts. We report a case of a 26-yr-old woman with both disorders and offer an alternative genetics-based etiology for the formation of choledochal cysts.

Adenomatous Polyposis Coli↗
PubMed1995

Choledochal cyst in pregnancy: a therapeutic dilemma.

BACKGROUND: Choledochal cysts occurring in pregnant women represent a diagnostic and therapeutic challenge to a broad spectrum of the medical profession. Not only is the association rare, but the clinical signs and symptoms are obscured by physiological changes that occur during pregnancy. As a result, diagnosis is often delayed until patients present with life-threatening complications. STUDY DESIGN: This report describes three cases of choledochal cysts occurring during pregnancy. RESULTS: Although the diagnosis was initially missed in two patients, delayed treatment was not associated with an adverse outcome. In a third patient, conservative management was complicated by rupture of the cyst which resulted in fetal loss and a protracted hospital course. Definitive cyst surgery resulted in a good long-term result in all three patients. CONCLUSIONS: Although choledochal cysts rarely occur in pregnancy, clinicians need to be aware of the condition, as delayed or inappropriate therapy may be catastrophic for both mother and child. Once the diagnosis is established, patients should be referred to specialized centers where treatment can be carefully planned, bearing in mind maternal and fetal well-being, as well as the likelihood of cyst-related complications both in the short- and long-term period. Excision with reconstruction is the procedure of choice to treat this type of cyst in nonpregnant patients. In pregnancy, however, a more conservative approach may have to be adopted until the second trimester or after delivery, when the surgical risk is lowest. Elective cesarean section should be undertaken in patients in whom the cyst has not been decompressed so as to avoid the complication of cystic rupture postpartum.

Abortion, Spontaneous↗
PubMed1990

Increased secondary bile acids in a choledochal cyst. Possible role in biliary metaplasia and carcinoma.

Choledochal cysts are uncommon congenital or acquired lesions of the biliary tree. The incidence of biliary tract carcinoma in patients with choledochal cysts is 5-35 times greater than that of the general population. Factors responsible for the increased risk of carcinoma are unknown. The case of a young woman who underwent excision of a choledochal cyst 16 years after initial diagnosis and treatment by choledochocystduodenostomy is reported. Metaplasia of the epithelial lining of the cyst was found in the resected specimen. The relative composition of bile acids in cyst contents was as follows: lithocholate, 2%; deoxycholate, 88%; chenodeoxycholate, 5%; and cholate, 5%. Virtually all bile acids were recovered in unconjugated form. In contrast, the bile acid composition of hepatic bile was as follows: lithocholate, 0%; deoxycholate, 34%; chenodeoxycholate, 43%; and cholate, 23%. Bile acids were fully conjugated. These data suggest that stasis of bile within choledochal cysts contributes to bacterial overgrowth and generation of unconjugated secondary bile acids.

Adenoma, Bile Duct↗
PubMed1991

Variation in management based on type of choledochal cyst.

The management of 23 patients treated for choledochal cysts at the Oregon Health Sciences University between 1969 and 1990 is reviewed. The median age was 27 years, with a range from 1 month to 90 years. Seventy-eight percent of patients presented with abdominal pain, and 35% were jaundiced. Three patients presented with cholangitis, two with cyst rupture, and one with recurrent pancreatitis. Nine patients had had previous biliary surgery. The diagnosis was made in all patients with ultrasound and/or cholangiography. Fifteen patients (65%) had type I cysts, 2 had a type II cyst, 5 (22%) had type III cysts, and 1 had a type IV cyst. Stones were present in four (17%) cysts, and all excised cysts were benign. Seventeen patients with type I and II choledochal cysts had complete cyst excision and choledochoenterostomy. Four of five patients with type III cysts had endoscopic cyst incision and drainage, while the fifth patient had transduodenal cyst excision and sphincteroplasty. The patient with a type IV cyst had extrahepatic cyst excision and choledochojejunostomy. There were no operative deaths. Two postoperative complications occurred: cholangitis and a prolonged ileus. All patients had resolution of their pain and jaundice. Two patients had late cholangitis. Cyst excision and choledochojejunostomy are the treatment of choice for types I and II choledochal cysts. Extrahepatic cyst excision and choledochojejunostomy may be adequate treatment for type IV cysts. Endoscopic incision and drainage is appropriate for selected patients with type III cysts.

Adolescent↗