ObjectiveTo investigate the feasibility and safety of subtotal hepatectomy with preservation of caudate lobe for extensive hepatolithiasis with atrophy of left and right hepatic lobe and obvious hypertrophy of caudate lobe.MethodThe clinicopathologic data of patient with hepatolithiasis whose left and right hepatic lobe atrophied and caudate lobe obviously hypertrophied admitted to the West China Hospital of Sichuan University in February 2020 were analyzed retrospectively.ResultsThe patient was in good general condition before the operation. The cardiopulmonary and kidney functions were normal. The liver function was Child-Pugh A grade, and the liver reserve function was good. The body surface area of the patient was 1.745 m2 and the standard liver volume was 1 235 mL. The volume of caudate lobe calculated by the 3D reconstruction of CT image was 735 mL, accounted for 59.5% of the standard liver volume. The patient was evaluated to be able to tolerate the operation. The patient successfully experienced the operation of subtotal hepatectomy with caudate lobe preservation. The postoperative liver function recovered well. The gastric tube was removed on the 4th day after the operation. The peritoneal drainage tube was removed on the 5th day after the operation. The patient was discharged on the 6th day after the operation. The postoperative pathological diagnosis: The intrahepatic bile duct was dilated with stones inside. A large number of inflammatory cell infiltrated around the bile duct. The fibrous tissue hyperplasia, small bile duct hyperplasia and inflammatory cell infiltration were observed in the portal area. The pathological changes were consistent with the changes of hepatolithiasis.ConclusionAccording to analysis results of this case, subtotal hepatectomy with preservation of caudate lobe is safe and feasible for hepatolithiasis patient with obvious atrophy of left and right hepatic lobe and obvious hypertrophy of caudate lobe.