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Signet-ring cell carcinoma (SRCC) is rare in the biliary system.We herein report a SRCC co-existing with adenocarcinoma arising in a choledochal cyst of the extrahepatic bile duct.A 52-year-old male was hospitalized due to jaundice and pruritus.Abdominal CT and MRCP showed a huge choledochal cyst and distal common bile duct cancer.The patient underwent a pancreaticoduodenectomy with extended lymph node dissection.A histologic study confirmed a SRCC co-existing with adenocarcinoma arising in a choledochal cyst.Due to serious side effects, he only underwent 2 cycles of chemotherapy after surgery.Four months after surgery, recurrence was observed in the bilioenteric anastomosis and he died six months after surgery.This patient is the first case of SRCC arising in a choledochal cyst of the extrahepatic bile duct in the world.