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AIM To understand the characteristics and investigate the diagnosis, treatment of intrahepatic biliarypapillomatosis.METHODS Two cases were confirmed operatively and pathologically, and the literature reviewed.RESULTS The causes of the disease are not clear, but the long-term irritations of stone and inflammationmay be considered. Despite being histologically benign, it has low-grade malignant potential, characterizedby multiple and diffusive manner, and has high rates of recurrence and malignant transformation. Jaundiceand cholangitis are the dominant manifestations. Imaging modalities are beneficial in it's diagnosis, and thescanning of choledochoscopy is of great value.CONCLUSION The management of the disease is difficult. Curative resection is the primary therapy.Curettage and drainage may lead to high recurrence. The chemotherapy and lasertherapy represent othermodalities in the treatment. Biliary papillomatosis may become a new indication for liver transplantation.
