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PubMed1985

Forme fruste choledochal cyst.

Four patients had the characteristic features of choledochal cyst except for the cystic component. All patients had stenosis of the distal common bile duct, a "long common channel" secondary to a proximal junction of the common bile and pancreatic ducts, cholecystitis and the classic pathological microscopic features of choledochal cyst in the wall of the common bile duct. Three children had coexisting intrahepatic duct cysts and/or stenosis and one had intrahepatic choledocholithiasis. The clinical presentations were cholangitis (2), pancreatitis (1) and biliary obstruction (1). In all cases the common bile duct was resected and biliary reconstruction was carried out by choledochojejunostomy (Roux-en-Y). Morbidity was minor except in one patient with ductal disease extending far into the intrahepatic ducts. This child developed an anastomotic stricture requiring revision of the anastomosis and long-term "U" tube stenting. Forme fruste choledochal cyst appears to be another variation in the spectrum of pancreaticobiliary malformations of choledochal cyst. Treatment is identical, that is, excision of all malformed ductal tissue.

Adolescent↗
PubMedDate not supplied

A case of carcinoma associated with the remained intrapancreatic biliary tract 17 years after the primary excision of a choledochal cyst.

Although the incidence of biliary carcinoma associated with choledochal cyst is high, there are extremely rare reports about cancer development in the remaining intrapancreatic biliary tract in patients who underwent primary excision of the choledochal cyst. The authors present a case of carcinoma associated with the remnant intrapancreatic biliary tract in a 39 year-old woman, 17 years after the initial excision of a choledochal cyst with Roux-en-Y hepaticojejunostomy. Long-term follow-up may be recommended even in patients who have undergone excisional surgery for choledochal cyst, because it is possible that cancer associated with the intrapancreatic biliary tract may develop.

Adenocarcinoma↗
PubMed1995

Prenatally diagnosed choledochal cysts: observation or early surgery?

The incidence of subhepatic cysts later confirmed as choledochal cyst has changed with the use of prenatal ultrasonography. A new group has emerged: the neonatal patient with an antenatal diagnosis. Optimal timing for cyst excision depends on variables such as gestational age, weight, associated conditions, biochemical liver alterations, development of complications, and the sonographic surveillance of size. The author's experience (1992) with an antenatally diagnosed choledochal cyst prompted this literature review. The present case showed an abnormal choledochopancreatic ductal junction, high amylase content, and a linear pattern of growth over time (2 mm/wk). Management consisted of cyst excision and bilio-enteric reconstruction. Fourteen cases (including the present one) have been reported in the world literature. All were females. Seventy-two percent of the ultrasound examinations were performed for dating purposes. Subhepatic cysts were identified at a mean gestational age of 26.9 weeks (range, 15 to 37 weeks). Excision and bilio-enteric reconstruction were performed at a mean age of 45 days (range, 9 hours to 6 months). Clinically, 50% of the babies were anicteric, 43% were jaundiced, and 7% had a palpable mass. Evidence of cyst growth was present in 56% of cases, and 60% had liver fibrosis that reverted to normal. The indications for surgery were jaundice (43%), cyst growth (21%), delayed HIDA excretion (7%), and elective (29%). Recommendations for managing the asymptomatic, anicteric neonate are discussed, entailing a rational approach based on growth potential, biochemical liver alterations, and the development of obstructive jaundice.

Adult↗
PubMed1982

Choledochal cysts in children: radiologic features.

Choledochal cysts, although uncommon, are found most frequently in children. Radiologic studies have always played an important role in diagnosis because of the nonspecific clinical features. The abdominal roentgenogram may show in the right upper abdomen a mass, the cystic nature of which can be demonstrated by intravenous urography, hepatic angiography, sonography, and computerized tomography. Large choledochal cysts will displace the duodenal loop inferiorly and to the left, as seen on upper gastrointestinal contrast studies. Oral cholecystography and intravenous cholangiography are currently not indicated because of the poor diagnostic yield and the significant toxicity of intravenous media in children. Operative cholangiography is desired by the surgeon for demonstration of the pathologic anatomy. Real-time sonography and cholescintigraphy with technetium Tc 99m iminodiacetic acid derivative allow specific preoperative diagnosis and will eliminate the need for more invasive studies, such as arteriography, endoscopic cholangiopancreatography, and percutaneous transhepatic cholangiography. The radiologic findings in six patients are summarized.

Angiography↗
PubMedDate not supplied

Choledochal cysts: evaluation with MR cholangiography.

BACKGROUND: To determine where magnetic resonance (MR) cholangiography can accurately demonstrate the presence, extent, and type of choledochal cysts. METHODS: Ten patients with sonographically suspected choledochal cysts were evaluated with a non-breath-hold MR cholangiography technique. The presence, extent, and type of choledochal cyst were determined. Visualization of the pancreatobiliary junction was recorded. MR cholangiographic findings were correlated with the gold standard of surgery in six patients, endoscopic retrograde cholangiography in two, and a 99mTc hepatobiliary scintigram in one. Three patients underwent intraoperative cholangiography. RESULTS: All MR cholangiograms were correlated with findings at surgery, endoscopic retrograde cholangiography, or 99mTc hepatobiliary scintigraphy. There were seven Todani type 1 and two Todani type 5 choledochal cysts (Caroli's disease). The extent of involvement was correctly demonstrated on all MR cholangiograms. The pancreatobiliary junction could not be identified in any of the cases. Calculi and sludge were correctly identified on the MR cross-sectional images in three patients but were not seen on MR cholangiograms in two. In one patient with an initially misinterpreted choledochal cyst, MR sectional images showed the typical appearance of a hydatid cyst, which was confirmed at surgery. CONCLUSION: MR cholangiography can be used to confirm the diagnosis of choledochal cysts and define the extent of involvement preoperatively. The pancreatobiliary junction, however, is difficult to visualize. Non-biliary cysts such as a hydatid cyst can mimic a choledochal cyst on the MR cholangiogram and should be correlated with the MR cross-sectional images to avoid misinterpretation.

Adolescent↗
PubMed1981

Management of adult choledochal cysts.

A review of the English literature reveals a total of 1,337 patients with choledochal cysts. Improved diagnostic techniques to visualize the biliary system are demonstrating an increasing number of unsuspected choledochal cysts in adult patients. Either choledochal cysts remain clinically silent until adulthood or may develop in later life. Experience is reported with adult patients having type I, II, III, and IV choledochal cysts. Type I cysts are preferably managed by excision but cyst anatomy may necessitate choledochoenteric drainage. Type II cysts are treated by excision except for those located within the pancreatic portion of the common bile duct. These are best managed by transduodenal cystoduodenostomy. The type III cyst (choledochocele) should be excised carefully, identifying and preserving the common bile and pancreatic ducts. Type IV cysts include a combination of any one of the first three types of cyst plus the presence of intrahepatic cyst or cysts. Treatment of these cysts is dictated by the type and location of the extrahepatic cyst. Since choledochal cysts are being recognized with increased frequency in adults, surgeons need to be aware of the diagnostic and treatment modalities available for each type of biliary cyst.

Adult↗
PubMed1993

Are secondary bile acids in choledochal cysts important as a risk factor in biliary tract carcinoma?

Since secondary bile acids have mutagenic potency and choledochal cyst patients with or without cyst-enterostomy have a high risk of developing bile duct carcinoma, we examined the hypothesis that secondary bile acids are elevated in the choledochal cyst and possibly cause biliary tract carcinoma. Eleven choledochal cyst patients with or without previous cyst-enterostomy or biliary tract carcinoma, seven patients with biliary tract carcinoma not associated with choledochal cyst and five patients with cholecystolithiasis were examined. Samples were directly needle aspirated from the cyst or by cannulating a tube into the common bile duct through the cystic duct. The concentrations of each bile acid in these samples were quantified by gas-liquid chromatography and compared. Neither the relative composition nor the absolute concentration of secondary bile acids (deoxycholic acid, lithocholic acid) elevated in patients with choledochal cyst compared with the values in the non-choledochal cyst patients with or without biliary tract carcinoma. The presence of biliary tract carcinoma or previous cyst-enterostomy did not affect the concentrations of secondary bile acids. The results suggest that the factor other than secondary bile acids can be primarily responsible for the high risk of bile duct carcinoma in patients with choledochal cyst.

Adult↗
PubMed1996

The clinical significance of gall-bladder non-visualization in cholescintigraphy of patients with choledochal cysts.

Intravenous radionuclide cholescintigraphy (IVRC) provides a very specific picture for choledochal cysts. However, the clinical significance of the non-visualization of the gall-bladder (GB) activity in these cases is unclear. In this study, we reviewed 27 patients with choledochal cysts who underwent IVRC within 7 days prior to operation and correlated the GB findings on IVRC with the histopathological results. In 18 of the 27 patients (66.7%), there was non-visualization of the GB at 4 h post injection. Among these, two had histopathological features of acute cholecystitis (AC), 11 had chronic cholecystitis (CC), and five had normal GBs. In the other nine patients with visualization of the GB, there were five cases of CC and four normal GBs. If we apply the finding of non-visualization of the GB at 4 h post injection as the criterion for the diagnosis of AC, the diagnostic accuracy was only 40.7% (11/27). We concluded that: (1) GB disease (AC and CC) was common (66.7%: 7.4% and 59.3% respectively) in choledochal cyst patients. (2) Non-visualization of the GB on IVRC did not necessarily indicate AC in choledochal cyst cases, and the diagnostic accuracy was low. (3) GB disease is not the only cause of GB non-visualization on IVRC. A huge choledochal cyst causing a mass or reservoir effect may be a cause of GB compression and result in non-visualization of the GB.

Child↗
PubMed1991

Type IVa choledochal cysts: surgical management and literature review.

Type IVa choledochal cysts with cylindrical dilatation of the intrahepatic ducts constitute a relatively less recognized variety of choledochal cysts, and differ from cystic dilatation of intrahepatic ducts in their clinical manifestations and response to treatment. Five patients with type IVa choledochal cysts and cylindrical dilatation of major intrahepatic ducts who underwent cyst excision and Roux-en-Y hepaticojejunostomy are reported. The duration of symptoms was less than 1 year in all patients. Palpable abdominal mass and abdominal pain were present in 3 patients. The traid of jaundice, abdominal pain and mass was present in only 1 patient. The intrahepatic dilatation regressed after excision of the extrahepatic cyst just below the hilum of the liver. The surgical technique is described and the need for excision of the cyst is emphasized.

Adolescent↗
PubMed1995

Does hyperamylasemia in choledochal cyst indicate true pancreatitis? An experimental study.

Patients with choledochal cyst often have repeated attacks of abdominal pain accompanied by hyperamylasemia, and they may be diagnosed as having acute pancreatitis. However, the attacks generally tend to subside in a short period by conservative treatment, and evidence of pancreatitis is rarely observed at the time of surgery. Choledochal cyst is commonly associated with pancreatobiliary maljunction, and high concentrations of pancreatic enzymes in bile are usually observed. When the bile duct pressure increases due to obstructive cholangitis, pancreatic enzymes in bile may regurgitate into the blood stream. Cholangiovenous reflux of amylase might cause hyperamylasemia. In order to investigate the mechanism of hyperamylasemia by cholangiovenous reflux, canine pancreatic juice or bile from a patient with choledochal cyst was injected into the obstructed common bile duct in dogs. The pancreatic enzymes in bile could readily enter into the blood stream at the pressure level of 15 mmHg or more in the bile duct. The peak amylase level in the thoracic lymph was observed to be more than 4 times higher than that in the blood serum, and the lymph flow during 30 minutes increased significantly from 8.1 to 20.4 ml at the bile duct pressure level of 20 mmHg. The reflux of amylase in bile into the blood stream via both the hepatic vein and thoracic duct might result in hyperamylasemia in the patients with choledochal cyst.

Acute Disease↗
PubMed1996

Presentation of choledochal cysts without intrabiliary communication on endoscopic retrograde cholangiopancreatography.

Choledochal cysts have generally been described to communicate with the biliary system. We recently saw three adult patients in whom we could not demonstrate biliary communication between the cyst and the biliary tree, despite a carefully performed endoscopic cholangiopancreatography (ERCP). These cases may represent a variant of type II choledochal cysts. During the 5-year period of review of ERCP records, 30 choledochal cysts were diagnosed, of which 3 (10%) were the noncommunicating cases described in this report.

Adult↗
PubMed2003

Choledochal cyst associated with portal hypertension.

Portal hypertension is a known complication of delayed presentation of choledochal cyst. In the literature, choledochal cyst presenting with portal hypertension has not been addressed adequately. The aetiology of portal hypertension in these cases has not been well studied, but it may be related to compression of the cyst over the portal vein, secondary biliary cirrhosis, or even extrahepatic portal venous thrombosis. We present our experience with four cases of choledochal cysts with associated portal hypertension over a 10-year period (1991-2001). Gastrointestinal bleeding, splenomegaly or incidental discovery of oesophageal varices led to the diagnosis of associated portal hypertension. Excision of choledochal cyst and bilioenteric bypass may not be feasible in those patients with massive collaterals in the region of porta with secondary portal hypertension. An initial approach of internal drainage (endoscopic/operative) of the cyst may allow regression of collaterals and subsequent safe excision of the cyst in this difficult group of patients. The liver function and histopathology dictated the final outcome.

Child↗
PubMed1997

Early and late results of excision of choledochal cysts.

BACKGROUND/PURPOSE: Reports on the late results of choledochal cyst excision with hepaticojejunostomy in children are relatively few. METHODS: Of the 84 patients who had choledochal cyst who came under our care, 79 have had definitive surgery, three are awaiting surgery, one is being observed with Caroli's disease, and the parents of one child have refused surgery. Thirty-eight patients treated decades ago had internal drainage procedures. Since 1972, 41 patients have had cyst excision with hepaticojejunostomy using a 40-cm Roux loop without an antireflux procedure. Early complications in those who underwent cyst excision with hepaticojejunostomy included anastomotic leak in three patients who required reoperation, cholangitis in two, and fluid collection in the gall-bladder bed that required no intervention in one. RESULTS: During a follow-up period ranging from 4 months to 17 years (mean, 8.5 years), anastomotic stricture, cholangitis, and intrahepatic stone formation developed in two children after being well for 8 years and over 11 years. These children required additional surgical procedures to overcome their problems. Asymptomatic intrahepatic stones 2 years after cyst excision with hepaticojejunostomy developed in a third child. There was no mortality in the entire group that underwent cyst excision and they are all enjoying a good quality of life. CONCLUSIONS: Careful, long-term follow-up is important in children who have choledochal cyst excision with hepaticojejunostomy.

Adolescent↗
PubMed2006

Aplasia of the dorsal pancreas and choledochal cyst.

The authors describe a rare case of choledochal cyst and aplasia of the dorsal pancreas complicated with chronic pancreatitis. A 9-year-old boy presented with obstructive jaundice. After biliary drainage using pericutaneous transhepatic gallbladder drainage (PTGBD) technique, the patient underwent choledochal cyst excision with Roux-en-Y hepaticojejunostomy. The association of choledochal cyst and aplasia of the dorsal pancreas with chronic pancreatitis has never been reported previously.

Child↗
PubMed1999

Laparoscope-assisted minimally invasive treatment for choledochal cyst.

The principle of treatment of choledochal cysts is total cyst excision with hepaticojejunostomy because of the high rate of associated malignancy of the biliary system. The authors used a minimally invasive laparoscopic procedure to treat a patient with nonmalignant choledochal cyst. Although a large median laparotomy is usually used for cyst excision and hepaticoenterostomy, laparoscope-assisted total cystectomy and hepaticojejunostomy were performed with minimal skin incision. To avoid gas embolism during dissection around the hepatic hilus the surgical procedure was divided into two stages: CO2 insufflation and abdominal lifting without pneumoperitoneum. This combination of procedures was as safe and technically adequate as conventional surgery. No abnormalities were observed in liver function, and the patient could sit up in bed the first day postoperatively. Thirteen days after surgery, he was discharged from the hospital uneventfully.

Adult↗
PubMed2005

Mixed type I and II choledochal cyst: a new clinical subtype?

Traditionally, choledochal cyst (CC) is classified into five types according to Todani's classification. Of 356 cases of CC treated at our institute since 1965, we experienced four cases of mixed type I and II choledochal cyst (MTCC). The incidence of MTCC was 1.1%. All four patients had a fusiform common bile duct (CBD) with a diverticulum originating from the mid-portion of the CBD. The cystic duct drained into the right side of the diverticulum. All had pancreaticobiliary malunion (PBMU). All presented with recurrent abdominal pain. MTCC was suggested on ultrasonography or computerized tomography and confirmed by endoscopic retrograde cholangiopancreatography (three cases) or magnetic resonance cholangiopancreatography (one case). Age at surgery ranged from 3 to 12 years. All had hepaticoenterostomy, and total excision of the CC/diverticulum/gallbladder. There were no complications and all cases are currently well. In all cases, histopathology showed that the wall of the diverticulum was identical to the CBD. Although the incidence of MTCC is very low, pediatric surgeons should be aware of it as a new subtype of CC. Based on our experiences and a review of the literature, we suggest that its etiology does not affect treatment because the surgical management of choice is the same as for type I CC.

Biliary Tract↗
PubMed2000

Choledochal cyst associated with duodenal obstruction.

The association between congenital duodenal obstruction and concomitant choledochal cyst has not been reported, although duodenal obstruction is known to be associated with many other anomalies. The authors describe 2 patients with choledochal cyst with duodenal obstruction. In 1 patient, a diverticulum type of choledochal cyst was found within an annular pancreas. Cyst excision, choledochojejunostomy, and side-to-side duodeno-duodenostomy were performed. The other patient showed separated duodenal atresia and other multiple anomalies including imperforate anus. A choledochal cyst was noted at the time of duodeno-duodenostomy and sigmoid colostomy. Cyst-enterostomy was performed at the age of 8 months, but the patient died of multiple anomalies. Intraoperative cholangiography indicated an anomalous pancreatobiliary ductal junction (APBDJ). In both patients the bile in the cyst contained high levels of amylase, suggesting the presence of an APBDJ. An APBDJ is considered to play an etiologic role in the development of the choledochal cysts associated with duodenal obstruction.

Choledochal Cyst↗
PubMed1990

Surgical treatment of congenital choledochal cyst.

20 cases of congenital choledochal cyst were operated by modified Lilly's method from 1980 to November 1985 in our Hospital. There were 4 males and 16 females. Their ages ranged from 50 days to 15 years with a mean of 5.1 years. All patients had preoperative ultrasonographic examination, barium meal radiography of the gastrointestinal tract or percutaneous transhepatic cholangiography. All had choledochal cystectomy with retention of the outer layer of the posterior wall of the cyst. We modified Lilly's method by injecting normal saline between the outer and inner layer of the choledochal cyst so that the outer layer could be isolated. Reconstruction of the biliary tract was then performed. 2 patients had choledochoduodenostomy and 18 patients had hepaticojejunostomy (1 end to end and 17 end to side). 19 patients had also had cholecystectomy. The post-operative course of the operation was found to be smooth and safe. Patients were followed up for a period from 6 months to 7 years. 3 patients had cirrhosis of liver, 2 of whom died within 8 months. The mortality rate in our series was 10%. The operative treatment and the problems of biliary reconstruction are discussed.

Adolescent↗