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Liver transplantation and whipple surgery combined with chemoradiotherapy for treatment of hilar cholangiocarcinoma in patients with primary sclerosing cholangitis

Cholangiocarcinoma (CC) is a mucin-producing adenocarcinoma that originates from the bile duct epithelium and is classified as intrahepatic, hilar, and peripheral according to its anatomic site of origin. Hilar CC is a fatal malignant tumor of the bile duct epithelium that is often diagnosed in advanced stages, and this leads to very short survival despite surgical intervention. The treatment of hilar CC usually includes bile duct resection and lymphadenectomy, with nearly 30% recurrence rates in the lungs and liver. However, the 5-year survival rate of patients after complete excision is less than 50%. CC is also considered a contraindication for orthotopic liver transplantation (OLT) because of the very poor survival rates of patients after OLT. Primary sclerosing cholangitis (PSC) is an inflammatory disorder of the biliary tree that is characterized by progressive fibrosis and stricturing of both intrahepatic and extrahepatic bile ducts that can lead to liver cirrhosis. Patients with PSC are at increased risk for the development of CC. Although the incidence rate of CC in patients with PSC is nearly 7%, a histological study of postmortem PSC livers or PSC patients who underwent OLT revealed a 30% rate of CC. OLT–Whipple surgery (en bloc total hepatectomy– pancreaticoduodenectomy–OLT) combined with radiotherapy has been reported as an approach to improve the survival rates of PSC patients with hilar CC. Herein, we describe 4 PSC patients affected with CC who were treated with liver transplantation and Whipple surgery combined with perioperative chemoradiotherapy. This study was approved by our local institutional review board.