Objective: To investigate the clinical features of intrahepatic biliary cystadenoma (IBCA), so as to provide reference for clinical use. Methods: The clinical articles concerning IBCA published over 15 years involving a total of 151 patients were collected, and diagnosis, treatment methods and outcomes of the patients were analyzed. Results: Of the 151 IBCA patients, 81 cases had preoperative misdiagnosis, so the misdiagnostic rate was 53.6%, which in the 23 reports of single case reached 91.3%. Of the patients, the average age was 49.5 years, number of females was higher than that of males (4.3:1), the general disease course was around 6 years, and the lesion was more frequently seen in the left liver than that in right liver (1.4:1). Both the imaging examinations and laboratory tests had less specificity in the early stage. After complete tumor resection, most cases had better outcomes and few cases had malignant transformation during follow-up. Conclusion: IBCA has no specific clinical manifestations, with high preoperative misdiagnostic rate, and intraoperative rapid frozen section procedure is important. Surgical resection is the main method of treatment. IBCA has a tendency to become malignant, so if possible, total resection of IBCA should be performed.