Complete Bile Duct Sequestration after Liver Transplantation, Caused by Ischemic-Type Biliary Lesions

Ischemic-type biliary lesions (ITBLs) are the most frequent cause of nonanastomotic biliary strictures in liver grafts, affecting about 2-19 % of patients after liver transplantation. ITBL is characterized by bile duct destruction, subsequent stricture formation, and sequestration. We report here the case of a patient affected by extremely severe ITBL, with sequestration and disintegration of the entire bile duct system, in which it was possible to extract the complete biliary tree endoscopically in a single piece. Histological examination revealed that all cells of the bile duct wall had been destroyed within 3 months after liver transplantation and replaced by connective tissue. Subsequently, biliary stricture formation occurred at the hepatic hilum, as well as the adjacent large bile ducts. It may be hypothesized that cellular rejection of small bile ducts leads to the vanishing bile duct syndrome, whereas cellular rejection of large bile ducts results in ITBL. The strictures were repeatedly dilated by endoscopic means, allowing successful control of stricture formation, as well as maintenance of liver function. At the time of writing, the grafted organ and the patient had survived for more than 3 years in good health. This is the first detailed report on a sequestration of the entire bile duct system caused by ITBL, successfully treated for several years by endoscopic means.

References

• 1 Hintze R E, Abou-Rebyeh H, Adler A. et al . Endoscopic therapy of ischemic-type biliary lesions (ITBL) in patients after orthotopic liver transplantation.  Z Gastroenterol. 1999;  37 13-20
• 2 Sanchez Urdazpal L, Gores G J, Ward E M. et al . Diagnostic features and clinical outcome of ischemic-type biliary complications after liver transplantation.  Hepatology. 1993;  17 605-609
• 3 Fisher A, Miller C H. Ischemic-type biliary strictures in liver allografts: the Achilles heel revisited?.  Hepatology. 1995;  21 589-591
• 4 Nakamura K, Murase N, Becich M J. et al . Liver allograft rejection in sensitized recipients: observations in a clinically relevant small animal model.  Am J Pathol. 1993;  142 1383-1391
• 5 Sanchez Urdazpal L, Batts K P, Gores G J. et al . Increased bile duct complications in liver transplantation across the ABO barrier.  Ann Surg. 1993;  218 152-158
• 6 Schlitt H J, Meier P N, Nashan B. et al . Reconstructive surgery for ischemic-type lesions at the bile duct bifurcation after liver transplantation.  Ann Surg. 1999;  229 137-145
• 7 Sebagh M, Farges O, Kalil A. et al . Sclerosing cholangitis following human orthotopic liver transplantation.  Am J Surg Pathol. 1995;  19 81-90
• 8 Johnson M W, Thompson P, Meehan A. et al . Internal biliary stenting in orthotopic liver transplantation.  Liver Transpl. 2000;  6 356-361
• 9 Davidson B R, Rai R, Kurzawinski T R. et al . Prospective randomized trial of end-to-end versus side-to-side biliary reconstruction after orthotopic liver transplantation.  Br J Surg. 1999;  86 447-452
• 10 Wiesner R H. Advances in diagnosis, prevention, and management of hepatic allograft rejection.  Clin Chem. 1994;  40 2174-2185
• 11 Baron P W, Heneghan M A, Suhocki P V. et al . Biliary stricture secondary to donor B-cell lymphoma after orthotopic liver transplantation.  Liver Transpl. 2001;  7 62-67
• 12 Seiler C A, Renner E L, Czerniak A. et al . Early acute cellular rejection: no effect on late hepatic allograft function in man.  Transpl Int. 1999;  12 195-201
• 13 Wiesner R H, Ludwig J, van Hoek B, Krom R AF. Current concepts in cell-mediated hepatic allograft rejection leading to ductopenia and liver failure.  Hepatology. 1991;  14 721-728
• 14 Tan C E, Vijayan V. New clues for the developing human biliary system at the porta hepatis.  J Hepatobiliary Pancreat Surg. 2001;  8 295-302
• 15 Mahajani R V, Cotler S J, Uzer M F. Efficacy of endoscopic management of anastomotic biliary strictures after hepatic transplantation.  Endoscopy. 2000;  32 943-949