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Erschienen in: Journal of Gastrointestinal Surgery 11/2015

01.11.2015 | Original Article

Surgical Management of Caroli’s Disease: Single Center Experience and Review of the Literature

verfasst von: Maitham A. Moslim, Ganesh Gunasekaran, David Vogt, Michael Cruise, Gareth Morris-Stiff

Erschienen in: Journal of Gastrointestinal Surgery | Ausgabe 11/2015

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Abstract

Background

Caroli’s disease is a rare congenital condition characterized by non-obstructive dilatation of intrahepatic ducts. In Caroli’s syndrome, there is additionally an associated congenital hepatic fibrosis.

Methods

With institutional review board approval, we identified all patients with Caroli’s disease and syndrome.

Results

Nine patients were identified, seven males and two females, with a median age of 40 years. Final pathological diagnoses included Caroli’s disease (n = 6) and Caroli’s syndrome (n = 3). Patients presented with deranged liver function, cholangitis, cholangiocarcinoma, abdominal pain, cirrhosis, or were diagnosed incidentally. Four patients underwent resection and two underwent liver transplantation. Of the resection group, two patients subsequently underwent transplantation for recurrent cholangitis due to anastomotic stricture in one patient and for end-stage liver disease in the other. All patients with Caroli’s syndrome underwent liver transplantation. Three patients died during follow-up at 26.2, 7.8, and 3 months post-diagnosis with recurrence of cholangiocarcinoma, liver failure, and metastatic cholangiocarcinoma, respectively. Six patients are alive with a median follow-up of 60 months since presentation (range = 10–134 months).

Conclusions

Caroli’s disease and syndrome have a varied presentation. Most individuals with Caroli’s disease may be adequately treated by resection, but transplantation is required for Caroli’s syndrome patients due to the associated hepatic fibrosis.
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Metadaten
Titel
Surgical Management of Caroli’s Disease: Single Center Experience and Review of the Literature
verfasst von
Maitham A. Moslim
Ganesh Gunasekaran
David Vogt
Michael Cruise
Gareth Morris-Stiff
Publikationsdatum
01.11.2015
Verlag
Springer US
Erschienen in
Journal of Gastrointestinal Surgery / Ausgabe 11/2015
Print ISSN: 1091-255X
Elektronische ISSN: 1873-4626
DOI
https://doi.org/10.1007/s11605-015-2918-9

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