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Bacterial cholangitis causing secondary sclerosing cholangitis: A case report
© ter Borg et al; licensee BioMed Central Ltd. 2002
Received: 28 March 2002
Accepted: 3 June 2002
Published: 3 June 2002
Although bacterial cholangitis is frequently mentioned as a cause of secondary sclerosing cholangitis, it appears to be extremely rare, with only one documented case ever reported.
A 48-year-old woman presented with an episode of acute biliary pancreatitis that was complicated by pancreatic abcess formation. After 3 months she had an episode of severe pyogenic (E. Coli) cholangitis that recurred over the subsequent 7 months on a further two occasions. Initially, cholangiography suggested the presence of extra-biliary intrahepatic abcesses while repeated investigations demonstrated development of multiple segmental biliary duct strictures. After maintenance antibiotic treatment was started, no episodes of cholangitis occurred over a 14-month period.
Sclerosing cholangitis can rapidly develop after an episode of bacterial cholangitis. Extra-biliary involvement of the hepatic parenchyma with abcess formation may be a risk factor for developing this rare but particularly severe complication.
Causes of biliary strictures
Primary sclerosing cholangitis
Previous biliary surgery
Rejection after liver transplantation
Hepatic arterial infusion of antineoplastic agents
Portal vein thrombosis / cavernoma
Parasitic biliary infection
Lymphoma / hepatic or metastatic carcinoma
This case clearly demonstrates the rapid development of sclerosing cholangitis after an episode of severe bacterial cholangitis. Although previous bacterial cholangitis is usually included in the differential diagnosis of sclerosing cholangitis, we have been unable to find more than a single reported case . Tanaka et al. described a patient who developed multiple intrahepatic biliary strictures after pyogenic cholangitis caused by Klebsiella and Enterococcus, that occurred as a complication of colonoscopic polypectomy. In both this and our case no obvious predisposing factors were identified. In particular, they were previously, healthy, immunocompetent patients without evidence to suggest pre-existing bile duct abnormalities or hepato-biliary disease. In our patient however, one can speculate about a role of the previous pancreatic abcesses in causing bacterial infestation of the biliary tree, as well as a possible role for the previous sphincterotomy in facilitating retrograde colonization of the biliary tree. Since the patient had a normal biliary anatomy except for the previous sphincterotomy, and had no signs or symptoms suggesting the presence of persistent infection of the biliary tree, we consider the presence of chronic cholangitis unlikely. The isolated bacterial organisms are commonly involved in bacterial cholangitis [4, 5]. At the time our patient presented with bacterial cholangitis, cholangiography and CT-imaging showed a picture suggesting the presence of multiple tiny parenchymal abcesses, communicating with the biliary tract. We speculate that this unusual extension of pyogenic cholangitis into the liver parenchyma may have played an etiological role in the subsequent development of biliary strictures, although another possibility is that the formation of these abcesses merely indicates the presence of severe cholangitis.
Our patient had repeated bouts of cholangitis once biliary strictures had developed. Antibiotic treatment and drainage led to rapid improvement on each occasion, but did not prevent new episodes. After maintenance antibiotic treatment was started, no further episodes of cholangitis occurred, and the patient remains in good clinical condition untill the moment of this writing. However, she seems to carry a considerable risk for developing future complications, including renewed bacterial cholangitis and development of secondary biliary cirrhosis.
Biliary strictures can develop as a complication of bacterial cholangitis. Extra-biliary hepatic involvement in the inflammatory process may be a risk factor for developing this rare complication.
Written consent was obtained from the patient for publication of the patient's details.
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