Background: Cholangiocarcinoma in patients with choledochoenterostomy has been a rare condition with few cases reported in the literature written in English, and there have been few pathological descriptions of this disease. However, it has recently been documented as a late complication of choledochoenteric anastomosis, and has become a serious issue today since choledochoenterostomy and sphincteroplasty for benign biliary lesions were frequently resorted to in the 1970s and 1980s. The authors here present a case of a 67-year-old woman who developed a cholangiocarcinoma 21 years after choledochoduodenostomy.
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