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Ex-vivo liver resection and liver autotransplantation offers a chance to cure extensive regional liver diseases which are otherwise deemed untreatable in selected cases. A 33-year-old male patient with advanced alveolar echinococcosis (AE) which invaded the right lobes and adjacent retrohepatic inferior vena cava, while the left lobes were free from lesion and had proliferated to over 1200 ml in size except for a 1 cm × 1 cm solid lesion in the segment Ⅳ. No extrahepatic metastases were found in this patient. A lesion with a size of 4300 ml and other small lesions in the right lobes were removed extracorporeally,and the retrohepatic vena cava was repaired, then the remaining AE-free left lobes (more than 700 ml in size) and the retrohepatic inferior vena cava were re-implanted in situ. A temporary end-to-end cavo-caval shunt with interposition of a blood vessel prosthesis and end-to-side portocaval shunt were established to keep the blood flow during the four-hour anhepatic phase. The patient was followed up for six months, and he had no signs of residual liver disease with good hepatic function.