Radical resection of recurrent cholangiocellular carcinoma (CCC) is rare. To date, only two patients have been reported to undergo major hepatectomy twice after pancreatoduodenectomy (PD) for metachronous CCC. This report describes a patient who underwent three metachronous radical resections of CCC with curative intent, consisting of PD followed by two hepatectomies, for cancer of the middle bile duct. A 60 year old male, who had undergone a distal gastrectomy for duodenal ulcer perforation in 23 years ago, underwent PD (Honjo method) for middle bile duct cancer (papillary adenocarcinoma) in 20 years ago at another hospital. In 12 years ago, he was referred to our hospital for further examinations of hepatic tumors and was diagnosed with CCC (S6, solitary, 4 cm). He underwent S6 subsegmentectomy of the liver. Pathological examination revealed a poorly differentiated CCC, considered a second primary cancer. In 6 years ago, subsequent follow up as an outpatient revealed repeated cholangitis, with CT showing a hilar mass caused by obstructive jaundice. He was diagnosed with a hilar cholangioma, and underwent hepatectomy and hepaticojejunostomy, which were safe despite surgical difficulties caused by severe adhesions and re-anastomosis. Pathological examination showed a welldifferentiated CCC, considered the third primary cancer. The lung metastasis was revealed in 28months after the last operation, however he has survived after partial lung resection without further metastasis
