Cases reported "Cholangitis, Sclerosing"

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1/23. Overlapping syndrome of autoimmune hepatitis and primary sclerosing cholangitis associated with pyoderma gangrenosum and ulcerative colitis.

    We describe the case of a 18-year-old male patient who first presented with decompensated cirrhosis, fever and generalized lymphadenopathy. He had abnormal results for liver biochemical tests, with a hepatitic-like picture and high titre of antinuclear antibodies. According to the scoring system proposed by the International Autoimmune hepatitis Group he had 'definite' autoimmune hepatitis and responded well to immunosuppressive treatment. One year later he developed pyoderma gangrenosum which was successfully treated with cyclosporine. Two years later he experienced bloody diarrhoea as a first presentation of ulcerative colitis. At that time both the cholestatic biochemical picture and the cholangiographic appearances of the biliary tree were consistent with primary sclerosing cholangitis. Despite the addition of azathioprine and ursodeoxycholic acid to his treatment regime he developed recurrent episodes of cholangitis and intractable pruritus for which he underwent successful liver transplantation.
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2/23. A case of sclerosing cholangitis managed by a percutaneous approach.

    In 1992, a 61-year-old man who complained of recurrent episodes of fever and jaundice was diagnosed as having sclerosing cholangitis. In the three years that followed, the clinical picture progressively worsened; and, in 1995, the patient was hospitalized again for biliary obstruction. A liver transplantation was excluded because of concomitant severe coronary heart disease. A percutaneous transhepatic cholangiogram showed several critical strictures of the intrahepatic biliary tree and a temporary internal-external biliary drainage was placed to relieve the obstruction. After 40 days, a two-step percutaneous biliary balloon dilation was performed followed by topical steroid treatment through the catheter. After 45 days, the catheter was removed and steroid treatment tapered orally. In the three years that followed, the patient was well. He experienced only about 1-2 episodes of ascending cholangitis per year requiring antimicrobial therapy. Laboratory analysis showed a gradual improvement in hepatic chemistry, serum bilirubin, and erythrocyte sedimentation rate (ESR). In our patient, the association of percutaneous balloon dilation and topical steroid treatment improved both the clinical and radiological picture, without significant side-effects. This approach should be considered a valuable and cost-effective option in primary sclerosing cholangitis, mainly for patients not eligible for liver transplantation.
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3/23. Hepatobiliary and pancreatic infections in AIDS: Part II.

    In the first part of our review, we discussed the general evaluation and clinical presentation of the various hepatic infections occurring in patients with AIDS. In addition, we focused on specific hepatic parenchymal infections. In this article, we will discuss the major clinical syndromes arising from opportunistic infections affecting the gallbladder (acalculous cholecystitis), biliary tree (AIDS-cholangiopathy), and pancreas (pancreatitis). acalculous cholecystitis can develop in patients with AIDS who have not experienced the severe precipitating physiologic stresses normally required in patients without AIDS. The most common presentation is with right upper quadrant (RUQ) pain and tenderness. The diagnosis is a clinical one since there is no standard test, other than surgery. cholecystectomy is the treatment of choice. The most common AIDS-associated infective complication of the biliary tree is AIDS-cholangiopathy. This is best viewed as a form of secondary sclerosing cholangitis resulting from a variety of opportunistic infections within the biliary tree. Affected persons present with RUQ pain and have marked elevations in the canalicular enzymes, alkaline phosphatase, and gamma-glutamyl transferase. Morphologic abnormalities are identified by endoscopic retrograde cholangiopancreatography. These include stricturing, dilatation, and beading of the biliary tract. Endoscopic sphincterotomy of the papilla of Vater may provide symptomatic relief for patients with papillary stenosis. opportunistic infections within the pancreas gland have been documented in both pre- and postmortem studies. However, the true incidence of pancreatitis related to infections is unknown. The presentation is similar to that of pancreatitis from other causes. A computerized tomogram of the abdomen is the investigation of choice. Tissue aspiration or biopsy of the pancreas is required to demonstrate the presence of an opportunistic infection. The management is usually supportive, as it is rare that a specific infection is identified and treated.
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4/23. Rapid progress of acute suppurative cholangitis to secondary sclerosing cholangitis sequentially followed-up by endoscopic retrograde cholangiography.

    A 66-year-old man was admitted to our hospital because of right hypochondralgia and fever after colonic polypectomy. Endoscopic examination revealed purulent bile excretion from the duodenal papilla orifice; based on this finding, acute suppurative cholangitis was diagnosed. An endoscopic retrograde cholangiography revealed no abnormality in the biliary tree. However, chronic cholestasis persisted, and endoscopic cholangiography performed 4 months later disclosed a beaded appearance of the intrahepatic bile ducts; this sign is a characteristic finding of sclerosing cholangitis. This is the first report of rapid progression of acute suppurative cholangitis to secondary sclerosing cholangitis sequentially followed-up by endoscopic retrograde cholangiography.
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5/23. Bile duct stenosis due to portal cavernomas: MR portography and MR cholangiopancreatography demonstration.

    We report two cases of bile duct stenosis due to portal cavernomas. Smooth stenoses were seen arising from both walls of the common bile duct on magnetic resonance (MR) cholangiopancreatography. On contrast-enhanced MR portography, peribiliary tortuous vessels were evident, indicating portal cavernomas. MR imaging can evaluate the biliary tree and portal systems noninvasively and was useful for evaluating this condition.
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6/23. Advanced cholangiocarcinoma in a patient with stage I primary sclerosing cholangitis.

    A 29-year-old woman presented with jaundice and fever in May 2001. cholangiography showed multiple strictures and beading of the biliary tree, with a large stricture in the common bile duct and marked dilatation of the hilar bile ducts. Typical cholangiography findings and elevated hepatobiliary enzymes suggested primary sclerosing cholangitis (PSC). At the same time, computed tomography detected a 2-cm tumor in the common bile duct, and angiography showed an encasement in the portal vein. Tumor markers, cytology, and biopsy were all negative for cancer. Although laparotomy showed a healthy liver and no lymph node metastasis was found, suggesting early-stage PSC and a low likelihood of accompanying cholangiocarcinoma (CCA) reported so far, the tumor in the resected common bile duct was subsequently diagnosed as CCA. Therefore, pancreatoduodenectomy was performed combined with partial resection of the portal trunk. histology also revealed invasion of the wall of the portal vein by cancer cells. The patient had a recurrence 5 months later and died 12 months after her operation. This is a rare case in which stage I PSC was complicated by advanced CCA.
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7/23. Primary sclerosing cholangitis.

    Primary sclerosing cholangitis (PSC) is an idiopathic, chronic cholestatic liver disease of uncertain etiopathogenesis commonly associated with inflammatory bowel disease (IBD) and is characterized by patchy inflammation of the biliary tree progressing to fibrosis and strictures. The natural history of PSC is highly variable but characteristically follows a progressive clinical course leading to biliary tree strictures, cholestasis, and choledocholithiasis. The course of the disease may be complicated by cholangitis, secondary biliary cirrhosis, liver failure, and cholangiocarcinoma. The diagnosis of PSC is based on typical cholangiographic findings, supported by nonspecific clinical signs and symptoms, cholestatic liver biochemical tests, and liver biopsy. Uncommon and usually clinically obvious secondary causes of sclerosing cholangitis are excluded before establishing the diagnosis of PSC. Therapeutic approaches that show promise include endoscopic therapy and ursodeoxycholic acid. The only accepted therapy for end-stage PSC that can improve long-term outcome is liver transplantation. The diagnosis of cholangiocarcinoma--often difficult and elusive--usually precludes liver transplantation because its prognosis is very poor.
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8/23. Pancreatic pseudotumor with sclerosing pancreato-cholangitis: is this a systemic disease?

    OBJECTIVE AND METHOD: Primary sclerosing cholangitis (PSC) is a disease that predominantly affects the biliary tree, although the pancreas may also be affected. A review of the presenting features of all patients given a diagnosis of PSC at a single center was conducted. The aim was to clarify the presentation of patients with pseudotumor of the pancreas in this patient population. RESULTS: Seventy-two patients were diagnosed with PSC either by ERCP (63/72 = 88%) or by liver biopsy (9/72 = 12%). The diagnosis of PSC was made following referral for abnormal liver tests (67%), jaundice (17%), and acute cholangitis (5%). Inflammatory bowel disease (IBD) (60%), non-insulin-dependent diabetes mellitus (NIDDM) (13%), thyroid disease (8%), and pancreatic disease (7%) were the major coexistent extrahepatic diseases. Three patients, all with marked weight loss, who presented with jaundice, abdominal pain, and/or diarrhea were found to have a pancreatic mass at first presentation. Clinical and radiological findings suggested pancreatic malignancy, and only later was advanced sclerosing cholangitis identified. The biopsy of the pancreas in two of these three patients revealed chronic pancreatitis. The long-term follow-up and good clinical response to medical therapy confirmed lack of pancreatic malignancy. These three patients all had other evidence of systemic involvement: submandibular gland fibrosis and urethral stricture in one, fibromuscular dysplasia of the renal artery in another, and retroperitoneal fibrosis in the third. None had IBD. CONCLUSION: Pancreatic pseudotumor with sclerosing pancreato-cholangitis may be a manifestation of a systemic disease characterized by nonmalignant strictures and multifocal fibroinflammatory processes, unlike classical PSC.
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9/23. Primary sclerosing cholangitis in which differential diagnosis from gallbladder carcinoma was difficult.

    We report a case of localized primary sclerosing cholangitis (PSC) which was difficult to distinguish from gallbladder carcinoma. A 75-year-old woman with elevated serum bilirubin was hospitalized and underwent endoscopic nasobiliary drainage (ENBD). There was no history of diseases such as gallbladder stone, pancreatitis, or ulcerative colitis. cholangiography through the ENBD tube showed localized stenosis of the common bile duct; the gallbladder could not be seen. Angiography showed no encasement of the hepatic artery. ultrasonography showed a tumor in the cystic duct, and the tumor had invaded the gallbladder and common bile duct. We diagnosed gallbladder carcinoma on radioimaging, and performed an S4aS5 subsegmentectomy of the liver and resection of the extrahepatic biliary tree. Pathologically, no malignant cells were detected, and fibrosis around bile ducts and infiltration of inflammatory cells into hepatic tissue were found. It is well known that PSC is sometimes difficult to differentially diagnose from cholangiocarcinoma. Our case is of high interest because ultrasonography showed findings suggestive of gallbladder carcinoma. It is therefore necessary to keep the possibility of PSC in mind for the diagnosis and treatment of such localized biliary stenosis.
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10/23. Orthotopic liver transplantation in an adult with situs inversus: an easy way to fit the liver.

    situs inversus (SI) is a rare congenital disorder with a complete mirror image of thoracic and abdominal organs. In adults with SI and decompensated cirrhosis experience with liver transplantation is limited. Orthotopic liver transplantation (OLT) in an adult with cirrhosis using a technique where the recipient liver was placed using a 90-degree rotation of the graft was previously reported by Klintmalm et al, however no other reports using this technique have been described. We report a case of a 41 year-old man with situs inversus and decompensated cirrhosis who successfully underwent OLT using this technique. The donor liver was rotated 90-degrees towards the left and easily fitted into the recipients'fossa with the left lobe pointing toward the left lower quadrant. The patient had an uneventful recovery and has been followed for 21 months without any complications. This technique has the advantage of preventing compromise of the size of the donor liver, permits an easy reconstruction of vascular and biliary tree and in this case was associated with an excellent outcome.
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