Purpose: There are sporadic reports of cancers developing in the remnant intrapancreatic bile duct tissues of patients with a history of primary choledochal cyst excision. The objective of this review is to study the clinical course of patients who develop subsequent biliary cancer originating from the remnant intrapancreatic bile ducts after cyst excision.
Methods: We describe a total of 17 cases (male:female 5:11; mean age 39.5 years), including the present case, from a review of the medical literature.
Results: Type I, type Iva, and unknown-type choledochal cysts according to the Todani classification were reported in nine, five, and three cases, respectively. The mean time to the development of subsequent cancer was 13.6 years. With the exception of one case, all of the cases (seven/eight cases) had elevated levels of serum CEA and/or CA19-9. Computed tomography was useful for detecting tumors (9/10 cases). Despite aggressive treatment, the cumulative survival rate after treatment was approximately 40 % at 1 year, with a mean survival duration of 12 months.
Conclusion: Cancer may develop up to 10 years after choledochal cyst excision, indicating the need for life-long follow-up in this patient population.
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