A 77-year-old woman underwent pancreatoduodenectomy with the diagnosis of biliary tract cancer. Postoperative immunohistochemical study showed endocrine cell carcinoma originating in the common bile duct. Systemic infusion of gemcitabine was started as adjuvant chemotherapy, however, 6 months after the operation, multiple liver and lymph node metastases were revealed by computed tomography. Then hepatic artery infusion with CPT-11 (40 mg/body) and CDDP (20 mg/body) were repeated every two weeks. Tumor markers normalized, and the size of both lymph nodes and liver tumors was remarkably decreased after 5 months. Endocrine cell carcinoma of the bile duct generally has a poor prognosis. This method could be a therapeutic option for recurrent endocrine cell carcinoma of the bile duct.

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