Article

Eosinophilic cholangiopathy with concurrent eosinophilic colitis in a patient with idiopathic hypereosinophilic syndrome

Division of Gastroenterology, University of Miami Leonard M. Miller School of Medicine, Miami, Florida 33136, USA.
European Journal of Gastroenterology & Hepatology (Impact Factor: 2.15). 07/2008; 20(6):574-7. DOI: 10.1097/MEG.0b013e3282f1cc11
Source: PubMed

ABSTRACT Eosinophilic cholangiopathy is a rare cholestatic disorder of unknown etiology. Simultaneous histologic documentation of eosinophilic involvement of the bile ducts and gastrointestinal tract has been reported previously in only a few well-documented cases. We report a 52-year-old man with a history of idiopathic hypereosinophilic syndrome who presented with acute diarrhea and cholestatic hepatitis. Colonoscopy revealed colitis, and a biopsy showed eosinophilic infiltrates in the colonic mucosa. Liver biopsy showed dense eosinophilic inflammation in the portal areas with bile duct damage. This case demonstrates the entity of hepatic involvement in idiopathic hypereosinophilic syndrome and the difficulties related to diagnosis and treatment.

0 Followers
 · 
60 Views
  • Source
    • "2007 26 M Fever, pruritis Yes CS NED, 6 mos 21 Sussman et al., [23] 2008 52 M Abdominal pain, pruritis Yes CS, AZT, UDCA NED 22 Raptou et al., [24] 2009 24 M Abdominal pain, fever Yes Aspiration, Albendazole NED 23 Current study, 2009 33 M Jaundice, pruritis No Roux-en-Y, CE, portal lymph- adenectomy, common bile excision NED, 13 mos "
    [Show abstract] [Hide abstract]
    ABSTRACT: Eosinophilic cholangitis (EC) is a rare benign disorder of the biliary tract which can cause biliary obstruction. Similar to other disease processes involving the bile ducts, this disorder can pose a difficult diagnostic challenge as it can mimic cholangiocarcinoma. A systematic search of the scientific literature was carried out using PubMed to access all publications related to EC. Search keywords that were utilized included "eosinophilic cholangitis," "etiology," "treatment," and "obstructive jaundice." Results. Twenty-three cases of EC have been reported. Nineteen patients (82.6%) who presented with EC remain disease-free; 15 of these 19 patients (78.9%) with followup time remain disease-free at a mean of 9.7 months (range, 2-24 months). EC is a rare form of biliary obstruction which can masquerade as a malignancy. Unlike cholangiocarcinoma, EC occurs more commonly in younger patients and in men. Most patients will require surgical treatment.
    HPB Surgery 11/2010; 2010:906496. DOI:10.1155/2010/906496
  • Source
    • "2007 26 M Fever, pruritis Yes CS NED, 6 mos 21 Sussman et al., [23] 2008 52 M Abdominal pain, pruritis Yes CS, AZT, UDCA NED 22 Raptou et al., [24] 2009 24 M Abdominal pain, fever Yes Aspiration, Albendazole NED 23 Current study, 2009 33 M Jaundice, pruritis No Roux-en-Y, CE, portal lymph- adenectomy, common bile excision NED, 13 mos "
    [Show abstract] [Hide abstract]
    ABSTRACT: Background. Eosinophilic cholangitis (EC) is a rare benign disorder of the biliary tract which can cause biliary obstruction. Similar to other disease processes involving the bile ducts, this disorder can pose a difficult diagnostic challenge as it can mimic cholangiocarcinoma. Methods. A systematic search of the scientific literature was carried out using PubMed to access all publications related to EC. Search keywords that were utilized included “eosinophilic cholangitis,” “etiology,” “treatment,” and “obstructive jaundice.” Results. Twenty-three cases of EC have been reported. Nineteen patients (82.6%) who presented with EC remain disease-free; 15 of these 19 patients (78.9%) with followup time remain disease-free at a mean of 9.7 months (range, 2–24 months). Conclusion. EC is a rare form of biliary obstruction which can masquerade as a malignancy. Unlike cholangiocarcinoma, EC occurs more commonly in younger patients and in men. Most patients will require surgical treatment.
    HPB Surgery 01/2010;
  • Source
    [Show abstract] [Hide abstract]
    ABSTRACT: Eosinophilic cholangiopathy is a rare condition characterized by eosinophilic infiltration of the biliary tract and causes sclerosing cholangitis. We report a patient with secondary sclerosing cholangitis with eosinophilic cholecystitis. A 46-year-old Japanese man was admitted to our hospital with jaundice. Computed tomography revealed dilatation of both the intrahepatic and extrahepatic bile ducts, diffuse thickening of the wall of the extrahepatic bile duct, and thickening of the gallbladder wall. Under the diagnosis of lower bile duct carcinoma, he underwent pylorus-preserving pancreatoduodenectomy and liver biopsy. On histopathological examination, conspicuous fibrosis was seen in the lower bile duct wall. In the gallbladder wall, marked eosinophilic infiltration was seen. Liver biopsy revealed mild portal fibrosis. He was diagnosed as definite eosinophilic cholecystitis with sclerosing cholangitis with unknown etiology. The possible etiology of sclerosing cholangitis was consequent fibrosis from previous eosinophilic infiltration in the bile duct. The clinicopathological findings of our case and a literature review indicated that eosinophilic cholangiopathy could cause a condition mimicking primary sclerosing cholangitis (PSC). Bile duct wall thickening in patients with eosinophilic cholangitis might be due to fibrosis of the bile duct wall. Eosinophilic cholangiopathy might be confused as PSC with eosinophilia.
    World Journal of Gastroenterology 04/2009; 15(11):1394-7. · 2.43 Impact Factor
Show more