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Rationale:  Choledochal cysts are a risk factor for the development of cholangiocarcinoma. Hence, complete surgical excision is the preferred treatment in most cases. However, cholangiocarcinoma still can develop from the remnant biliary system after surgical excision. Signet-ring cell carcinoma is a rare type of cancer of the biliary system, and the occurrence of signet-ring cell carcinoma after surgical excision of choledochal cysts has not been reported in the English literature to date.    Patient concerns:  We report a case of a 32-year-old woman who presented with a 1-month history of abdominal pain,obstructive jaundice, itching, and fever. The patient had undergone choledochal cyst excision and Roux-en-Y hepatico-jejunostomy 25 years previously and had now developed signet-ring cell carcinoma along with cholangiocarcinoma at the anastomotic site.    Diagnoses:  signet-ring cell carcinoma along with cholangiocarcinoma.    Interventions:  Interventions included laparotomy with evacuation,blood transfusion,and other adjuvant therapy.    Outcomes:  The patient died five months later.    Lessons:  Surgery is the best treatment for CCCs, and the surgeon should try to remove as much as of the bile duct cyst as possible.

Occurrence of signet-ring cell carcinoma with cholangiocarcinoma 25 years after choledochal cyst excision