Resolution of tuberculous biliary stricture after medical therapy

Tuberculosis (TB) is a very rare cause of biliary stricturethat is difficult to diagnose and usually requires surgicalintervention in order to rule out underlying malignantetiology.We report a 56-year-old man presented withjaundice,weight loss and poor appetite.Initial work upshowed the dilated biliary system secondary to distalcommon bile duct stricture.Investigations to definethe etiology of this stricture showed inconclusive brushcytology with absent abdominal masses and lymph nodesbut enlarged mediastinal lymph nodes.Biopsy fromthese lymph nodes showed a non-caseating epitheliodgranuloma with negative acid fast bacilli(AFB)stain.The patient had a dramatic response to empirical anti-tuberculosis therapy.Six weeks later,culture from lymphnodes was positive for Mycobacterium tuberculosis.Three months later,follow-up cholangiogram showedcomplete resolution of the stricture with normalizationof liver enzymes 6 mo after starting anti-tuberculosistherapy.Treatment was continued for 12 mo and thepatient had a normal life with normal liver enzymes andregression of the mediastinal lymph nodes at the timewhen he was reported in this paper.Although 16 cases of tuberculous biliary stricture areavailable in the English literature,up to our knowledge,this is the second published report of tuberculous biliarystricture,which resolved completely after medicaltherapy alone and the second reported case from theMiddle East.This report emphasizes the importance ofkeeping TB as a possibility of biliary stricture in this partof the world.     

Asymptomatic adenomyoma of the common hepatic duct discovered during a medical checkup: report of a case

Although adenomyoma of the gallbladder is not so rare, adenomyoma arising in the bile duct is extremely rare. We herein report a case of partial biliary obstruction due to adenomyoma of the common hepatic duct. A 64-year-old woman was referred to the 1st Department of Surgery, Niigata University School of Medicine for surgical intervention, in whom a biliary stricture in the common hepatic duct was discovered incidentally during a medical check-up for hyperamylasemia in an affiliated hospital. She was asymptomatic without jaundice. Based on her past history of abdominal contusion, the insidious presentation and the smooth biliary stricture on cholangiography, a diagnosis of benign biliary stricture secondary to blunt abdominal trauma was made. A suspicion of malignancy, however, could not be ruled out. Subsequently, she underwent a resection of the extrahepatic bile duct with lymph node dissection in the hepatoduodenal ligament. The patient's postoperative course was uneventful. She was discharged 18 days after the operation. Histology of the resected specimen confirmed the diagnosis of adenomyoma in the common hepatic duct. Clinicians should note to avoid overlooking or misdiagnosing that adenomyoma is a cause of biliary obstruction, although extremely rare.     

Spontaneous rupture of intrahepatic biliary ducts with biliary peritonitis.

Spontaneous rupture of intrahepatic biliary ducts is a rare cause of acute abdomen due to biliary peritonitis. We report a 92-year-old woman with 48-h history of upper abdominal pain, nausea and vomiting and peritoneal signs. CT scan showed free fluid in the abdomen and mild dilatation of the common bile duct. Exploratory laparotomy showed bile in the abdominal cavity with leak-age from a ruptured bile duct radicle in segment 3, as confirmed on intraoperative cholangiography. She underwent cholecystectomy, choledochotomy with removal of gallstones, repair of the perforation with primary suture and placement of a T-tube. She had an uneventful recovery.     

Tuberculous lymphadenitis as a cause of obstructive jaundice： A case report and literature review

Obstructive jaundice secondary to tuberculosis （TB） is extremely rare. It can be caused by TB enlargement of the head of the pancreas, TB lymphadenitis, TB stricture of the biliary tree, or a TB mass of the retroperitoneum. A 29-year-old man with no previous history of TB presented with abdominal pain, obstructive jaundice, malaise and weight loss. Ultrasonography （US）, computer tomography （CT） scan and endoscopic retrograde cholangiopancreatography （ERCP） were suggestive of a stenosis of the distal common bile duct （CBD） caused by a mass in the posterior head of the pancreas. Tumor markers, CEA and CA19-9 were within normal limits. At operation, an enlarged, centrally caseous lymph node of the posterior head of the pancreas was found, causing inflammatory stenosis and a fistula with the distal CBD. The lymph node was removed and the bile duct resected and anastomosed with the Roux-en Y jejunal limb. Histology and PCR based-assay confirmed tuberculous lymphadenitis. After an uneventful postoperative recovery, the patient was treated with anti-tuberculous medication and remained well 2.5 years later. Though obstructive jaundice secondary to tuberculous lymphadenitis is rare, abdominal TB should be considered as a differential diagnosis in immunocompromised patients and in TB endemic areas. Any stenosis or fistulation into the CBD should also be taken into consideration, and biliary bypass surgery be performed to both relieve jaundice and prevent further stricture.     

Biliary tuberculosis causing cicatricial stenosis after oral anti-tuberculosis therapy

A 36-year-old Philippine woman presented with dark urine and yellow sclera. Endoscopic retrograde cholangiopancreatography (ERCP) confirmed dilatation of the intrahepatic bile ducts and also showed an irregular stricture of the common hepatic duct at the liver hilum. Histological examination of biopsies from the bile duct revealed epithelioid cell granulomas and caseous necrosis. Tubercle bacilli were then detected on polymerase chain reaction (PCR) testing of the bile, giving the diagnosis of biliary tuberculosis. Although microbiological cure was confirmed, the patient developed cicatricial stenosis of the hepatic duct. She underwent repeated treatments with endoscopic biliary drainage (EBD) tubes and percutaneous transhepatic biliary drainage (PTBD) tubes, and the stenosis was corrected after 6 years. We present a case of tuberculous biliary stricture, a condition that requires careful differentiation from the more common malignancies and needs long-term follow-up due to the risk of posttreatment cicatricial stenosis, although it is rare.     

Biliary tuberculosis causing cicatricial stenosis after oral anti-tuberculosis therapy

A 36-year-old Philippine woman presented with dark urine and yellow sclera. Endoscopic retrograde cholangiopancreatography (ERCP) confirmed dilatation of the intrahepatic bile ducts and also showed an irregular stricture of the common hepatic duct at the liver hilum. Histological examination of biopsies from the bile duct revealed epithelioid cell granulomas and caseous necrosis. Tubercle bacilli were then detected on polymerase chain reaction (PCR) testing of the bile, giving the diagnosis of biliary tuberculosis. Although microbiological cure was confirmed, the patient developed cicatricial stenosis of the hepatic duct. She underwent repeated treatments with endoscopic biliary drainage (EBD) tubes and percutaneous transhepatic biliary drainage (PTBD) tubes, and the stenosis was corrected after 6 years. We present a case of tuberculous biliary stricture, a condition that requires careful differentiation from the more common malignancies and needs long-term follow-up due to the risk of posttreatment cicatricial stenosis, although it is rare.     

A case of percutaneous and transpapillary placements of expandable metallic stents in a patient with cholangiocarcinoma at the hilum of the liver

A 76-year-old woman was admitted with obstructive jaundice. US and MR cholangiopancreatography (MRCP) revealed an inoperative cholangiocarcinoma, 3 cm in diameter at the hilum of the liver, the obstruction of the hepatic duct bifurcation and the separation of bilateral hepatic bile ducts. Percutaneous transhepatic biliary drainage (PTBD) was performed from bilateral hepatic bile ducts. The right PTBD tube was spontaneously extubated. We could not succeed in performing internal biliary drainage across the hilar malignant stricture from a left hepatic bile duct, because of bad angulation. Transpapillary insertion into the common bile duct (CBD) was extremely difficult due to the collapse of the CBD. Endoscopic sphincterotomy (EST) after precutting method was performed. Although we performed the ballooned dilatation of malignant stricture and the insertion of a self-expandable metallic stent (EMS) into a right hepatic bile duct transpapillary. After dilatation of the hilar malignant stricture by the initial EMS, we inserted a guidewire into the CBD through the wire mesh of a stent from the left PTBD tube. We could insert the second EMS from a left hepatic bile duct to the CBD transhepatically, using a dilator and a dilating balloon. Finally, we performed the ballooned dilatation from bilateral hepatic bile ducts to the CBD transpapillary. She was discharged after bilateral internal biliary drainages, successfully.     

Self-expanding metallic stent for benign biliary strictures: seven-year follow-up

We report two cases of benign biliary strictures managed by metallic stent placement because the patients refused surgical repair. One patient is a 67-year-old man who had a stricture of the right hepatic duct and a bile leak following a cholecystectomy. The second patient is a 50-year-old man who had a stricture of the left hepatic duct following a right hepatic lobectomy for hepatolithiasis. For treatment of these bile duct strictures, a Gianturco-Rosch Z stent was placed in the former case and a Wallstent in the latter. Luminal patency of the stent was maintained for 7 years in the former case but in the latter, luminal stenosis of the stent was induced soon after placement. However, in the former, bile stasis in the left hepatic duct system, which emptied into the side of the stent, gradually developed without signs or symptoms of cholangitis or biliary obstruction. Therefore, the use of metallic stents for benign biliary stricture remains controversial. However, if metallic stent placement is the only therapeutic option, it is necessary to maintain bile flow not only through the stent but also in the bile ducts which flow into the side of the stent.     

Placement of removable metal biliary stent in post-orthotopic liver transplantation anastomotic stricture

Postoperative biliary strictures are the most common cause of benign biliary stricture in Western countries, secondary to either operative injury or bile duct anastomotic stricture following orthotopic liver transplantation(OLT).Surgery or endoscopic interventions are the mainstay of treatment for benign biliary strictures.We aim to report the outcome of 2 patients with refractory anastomotic biliary stricture post-OLT,who had successful temporary placement of a prototype removable covered self-expandable m...     

Pseudoaneurysm following laparoscopic cholecystectomy

BACKGROUND:Laparoscopic cholecystectomy(LC)is the operation of choice for removal of the gallbladder. Unrecognized bile duct injuries present with biliary peritonitis and systemic sepsis.Bile has been shown to cause damage to the vascular wall and therefore delay the healing of injured arteries leading to pseudoaneurysm formation.Failure to deal with bile leak and secondary infection may result in pseudoaneurysm formation. This study was to report the incidence and outcomes of pseudoaneurysm in patients with bile leak following LC referred to our hospital. METHODS:A retrospective analysis of our prospectively maintained liver database using pseudoaneurysm, bile leak and bile duct injury following laparoscopic cholecystectomy from January 2000 to December 2005 was performed. RESULTS:A total of 86 cases were referred with bile duct injury and bile leak following LC and of these,4 patients (4.5%)developed hepatic artery pseudoaneurysm(HAP) presenting with haemobilia in 3 and massive intra- abdominal bleed in 1.Selective visceral angiography confirmed pseudoaneurysm of the right hepatic artery in 2 cases,cystic artery stump in one and an intact but ectatic hepatic artery with surgical clips closely applied to the right hepatic artery at the origin of the cystic artery in the fourth case.Effective hemostasis was achieved in 3 patients with coil embolization and the fourth patient required emergency laparotomy for severe bleeding and hemodynamic instability due to a ruptured right hepatic artery.Of the 3 patients treated with coil embolization, 2 developed late strictures of the common hepatic duct. . (CHD)requiring hepatico-jejunostomy and one developed a stricture of left hepatic duct.All the 4 patients are alive at a median follow up of 17 months(range 1 to 65)with normal liver function tests. CONCLUSIONS:HAP is a rare and potentially life- threatening complication of LC.Biloma and subsequent infection are reported to be associated with pseudoaneurysm formation.Late duct stricture is common either due to unrecognized injury at LC or secondary to ischemia after embolization.     

Cholestasis as presenting symptom of portal cavernoma

A 51-year-old man with a history of portal vein thrombosis, was examined because of elevated liver tests and a tumoral mass in the liver hilus. Computed tomography (CT) scan and magnetic resonance imaging confirmed the portal vein thrombosis and an infiltrating mass in the porta hepatis with compression on the common bile duct. Endoscopic retrograde cholangiography showed an irregular narrowing of the mid-part of the common bile duct. The patient was referred for explorative laparotomy, which revealed a hypervascular mass in the liver hilus surrounded by many blood vessels. The diagnosis of portal cavernoma was made. Further haematological examination for the cause of portal vein thrombosis revealed an anti-phospholipid syndrome as well as myeloproliferative disease. Oral anticoagulant treatment is started. In conclusion, we report a case of biliary stricture due to portal vein thrombosis and cavernoma (portal biliopathy) which was not diagnosed preoperatively. Biliary strictures associated with portal vein thrombosis are due to extrinsic compression by collaterals and can also be induced by ischemic injury secondary to venous and arterial thrombosis of the choledochal vascular plexus.     

Biliary tuberculosis mimicking cholangiocarcinoma: treatment with metallic biliary endoprothesis

A 58-yr-old patient who presented with obstructive jaundice was evaluated with ultrasonography (US), computed tomography (CT), and percutaneous transhepatic cholangiography (PTC). Diffuse irregular stenosis of the extrahepatic bile ducts and periductal ill-defined soft tissue density along the hepatoduodenal ligament was determined. The patient was originally misdiagnosed with cholangiocarcinoma and, because the extent of disease process made surgical bypass impossible, was treated with a percutaneously inserted metallic stent. Histopathological examination of the endoluminal biopsy revealed ductal tuberculosis (TB). Most of the previous reports in the literature indicated that biliary obstruction was due to enlarged tuberculous lymph nodes compressing the bile duct. To our knowledge, only three cases of biliary stricture due to tuberculous involvement of the bile ducts were reported previously. This case illustrates the importance of tissue diagnosis in all cases of obstructive jaundice to avoid missing rare but curable diseases.     

A case of spontaneous biloma complicated with choledocholithiasis and chronic cholecystitis]

A biloma is an encapsulated bile collection outside the biliary tree. Most cases of biloma are caused by iatrogenic injury or trauma. Intrahepatic rupture of the biliary tree due to nontraumatic cause is a rare event. A 68- year-old man was admitted because of abdominal pain and fever. He had no past history of abdominal surgery, instrumentation or trauma. Computed tomography (CT) scan and magnetic resonance cholangiopancreatography (MRCP) demonstrated a large subcapsular fluid collection in the right liver associated with choledocholithiasis and cholecystitis. Biloma was confirmed by sono-guided percutaneous needle aspiration and was drained through a pigtail catheter. After the successful treatment by percutaneous drainage and endoscopic sphincterotomy, the patient recovered. Here, we report an uncommon case of spontaneous biloma formation in association with choledocholithiasis with a review of literatures.     

Computed tomography virtual endoscopy with angiographic imaging for the treatment of type IV-A choledochal cyst

Type IV-A choledochal cysts (CCs) are a congenital biliary anomaly which involve dilatation of the extrahepatic and intrahepatic bile ducts. We present the case of a 30-year-old woman with type IV-A CC, on whom three-dimensional computed tomography (3D CT) and virtual endoscopy were performed. 3D CT revealed partial dilatation in the posterior branch of the intrahepatic bile duct and a relative stricture between it and the extrahepatic bile duct. Virtual endoscopy showed that this stricture was membrane-like and separated from the surrounding blood vessels. Based on these image findings, complete cyst resection, bile duct plasty for the stricture, and hepaticojejunostomy were safely performed. To the best of our knowledge, there are no reports of imaging by virtual endoscopy of the biliary tract which show the surrounding blood vessels running along the bile duct.     

Follicular cholangitis mimicking cholangiocarcinoma treated with right hepatectomy: A case report and review of published works

Follicular cholangitis is a new, rare disease that causes severe biliary stricture. We herein describe the findings from a resected case of follicular cholangitis, suggesting a distinct disease entity that causes benign biliary stricture. A 60‐year‐old man who was referred to our hospital due to elevated γ‐glutamyl transpeptidase levels and dilatation of the B8 bile duct. Although bile juice cytology and bile duct brushing cytology showed no malignancy, the dilatation was progressive. Therefore, right hepatectomy combined with caudate lobectomy was carried out on suspicion of intrahepatic cholangiocarcinoma. The wall of the resected bile duct was markedly thickened due to severe fibrosis under the mucosal layer. Histology of the mucosal epithelium indicated no malignancy. Infiltration of plasma cells characterized by remarkable formation of lymphoid follicles with germinal centers was observed around the bile ducts. The patient was diagnosed with follicular cholangitis based on histological findings. We thus observed a rare case of follicular cholangitis. This case and review of published reports suggest that, despite its rarity, follicular cholangitis should be considered at the differential diagnosis of biliary stricture. This case report could contribute to a better understanding of how to address this disease.     

An unusual cause of biliary stricture in a patient with neurofibromatosis type 1

The majority of biliary strictures occur as a consequence of iatrogenic injury to the extrahepatic biliary tract, with more than 80% following cholecystectomy. The laparoscopic era has led to heightened awareness of this problem. The occurrence of an iatrogenic stricture can be particularly devastating to both patient and surgeon. The literature highlights a number of factors involved in the aetiology of such traumatic stricture formation. We report an unusual case of a Bismuth 2 stricture of the proximal common hepatic duct,occurring in a patient with type 1 neurofibromatosis, following an iatrogenic bile duct injury that occurred during a laparoscopic cholecystectomy. Histological examination of the strictured region of bile duct removed at surgery demonstrated multiple neurofibromas of varying sizes present in the submucosa. Neurofibromatosis type 1 (von Recklinghausen disease) affects the gastrointestinal tract in up to 25% of cases, and in such cases is characterized by multiple submucosal neurofibromas. We believe this is the first reported case of a biliary stricture in a patient with neurofibromatosis type 1, which appeared to be as a consequence of neurofibromas in the submucosa of the bile duct.     

Eosinophilic Cholangitis—A Challenging Diagnosis of Benign Biliary Stricture: A Case Report

When confronting a biliary stricture, both benign and malignant etiologies must be carefully considered as a variety of benign biliary strictures can masquerade as hilar cholangiocarcinoma (CCA). Therefore, patients could undergo a major surgery despite the possibility of a benign biliary disease. Approximately 15% to 24% of patients undergoing surgical resection for suspected biliary malignancy will have benign pathology. Eosinophilic cholangitis (EC) is a rare benign disorder of the biliary tract, which can cause obstructive jaundice and can pose a difficult diagnostic task.We present a rare case of a young woman who was referred to our hospital with obstructive painless jaundice due to a biliary stricture at the confluence of the hepatic bile ducts, with a provisional diagnosis of cholangiocarcinoma.Though, during her work up she was found to have EC, an extremely rare benign cause of biliary stricture, which is characterized by a dense eosinophilic infiltration of the biliary tree causing stricturing, fibrosis, and obstruction and which is reversible with short-term high-dose steroids.Despite its rarity, EC should be taken into consideration when imaging modalities demonstrate a biliary stricture, especially if preoperative diagnosis of malignancy cannot be made, in the setting of peripheral eosinophilia and the absence of cardinal symptoms of malignancy.     

Spontaneous perforation of right hepatic duct in a child with choledochal cyst

A 5-year-old girl with choledochal cyst (Alonso Lej, type I) and spontaneous perforation of the right hepatic duct during an attack of acute pancreatitis was treated by two-stage surgery. Biliary panperitonitis was diagnosed by abdominal paracentesis, bilirubin and amylase levels in the collected fluid being 41.3 mg/dl and 1812 Somogyi U, respectively. The initial surgery involved simple peritoneal drainage and decompression of the bile duct, followed by removal of a choledochal cyst and the common hepatic bile duct, including the biliary stricture of the left hepatic duct, and biliary reconstruction 5 months later. Cholangiogram after the initial surgery demonstrated pancreaticobiliary maljunction and stricture in the left hepatic duct and the common hepatic duct. Regurgitation of pancreatic juice into the bile duct was shown by the extremely high amylase level (90100 Somogyi U) in the bile drained through a T-tube. The patient is currently well, with normal liver and pancreatic function tests. The experience in this patient is significant in that it should be known that spontaneous perforation of the bile duct may occur at the right hepatic duct in choledochal cyst, causing biliary peritonitis.     

Diagnosis of cholangiocarcinoma

Cholangiocarcinoma is suspected based on signs of biliary obstruction, abnormal liver function tests, elevated tumor markers (carbohydrate antigen 19-9 and carcinoembryonic antigen), and ultrasonography showing a bile stricture or a mass, especially in intrahepatic cholangiocarcinoma. Magnetic resonance imaging (MRI) or computed tomography (CT) is performed for the diagnosis and staging of cholangiocarcinomas. However, differentiation of an intraductal cholangiocarcinoma from a hypovascular metastasis is limited at imaging. Therefore, reasonable exclusion of an extrahepatic primary tumor should be performed. Differentiating between benign and malignant bile duct stricture is also difficult, except when metastases are observed. The sensitivity of fluorodeoxyglucose positron emission tomography is limited in small, infiltrative, and mucinous cholangiocarcinomas. When the diagnosis of a biliary stenosis remains indeterminate at MRI or CT, endoscopic imaging (endoscopic or intraductal ultrasound, cholangioscopy, or optical coherence tomography) and tissue sampling should be carried out. Tissue sampling has a high specificity for diagnosing malignant biliary strictures, but sensitivity is low. The diagnosis of cholangiocarcinoma is particularly challenging in patients with primary sclerosing cholangitis. These patients should be followed with yearly tumor markers, CT, or MRI. In the case of dominant stricture, histological or cytological confirmation of cholangiocarcinoma should be obtained. More studies are needed to compare the accuracy of the various imaging methods, especially the new intraductal methods, and the imaging features of malignancy should be standardized.     

Follicular cholangitis: another cause of benign biliary stricture

The case of a 57-year-old woman with a benign biliary stricture is described. Radiological examinations showed granularly elevated lesions in the common hepatic duct and severe stenosis at the hepatic hilum. Under a tentative diagnosis of hepatic hilar bile duct cancer, the patient underwent extended right hepatectomy with bile duct resection. However, pathological and immunohistochemical studies revealed a benign stricture with remarkable formation of lymph follicles with germinal centers, which could be termed "follicular cholangitis". This is the first case report of this uncommon presentation of inflammatory changes of the bile duct. Its etiology was unknown, and more investigation is encouraged to clarify its relationship to systemic disorders and malignancies. As difficulty remains in discriminating between benign and malignant strictures by nonsurgical modalities at present, surgical resection without complications provides a feasible approach to such a benign lesion.