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Biliary cystadenoma (BCA) is a rare benign tumor, accounting for only 5% of reported cystic lesions of the liver. Given its potential for malignancy and high rate of recurrence, surgical resection is the preferred treatment. Therefore, early and accurate preoperative diagnosis is critical to the choice of treatment. We here report the first male case of BCA in our hospital, diagnosed by our team and confirmed by pathological biopsy. This article aims to improve the understanding of this disease and help make a correct diagnosis to