Hyperacute rejection is rare among ABO-compatible liver transplantations. The mechanism is donor preformed antibodies causing graft loss within a few days. Herein, we have described a case of an ABO-compatible liver transplantation that underwent hyperacute rejection, needing retransplantation for treatment. A 27-year-old man of blood group A positive who displayed fulminant hepatic failure due to hepatitis B (in agreement with the O'Grady criteria), received an ABO-compatible graft. He developed significant asthenia, fever, hypotension, oliguria, and coagulopathy. Ultrasonography revealed total thrombosis of the portal vein and absence of dilatation of bile ducts. The patient was priorized for retransplantation and underwent a good subsequent evolution. On anatomopathologic exam the explant revealed thrombosis of the intrahepatic branches of the portal vein with venous and ischemic infarcts compatible with a diagnosis of hyperacute rejection. The clinical findings of hyperacute rejection were characterized by progressive elevation of bilirubin and thrombocytopenia associated with signs of hepatic failure during the first days after transplantation. In this case, the histological exam was compatible with hyperacute rejection, excluding the diagnoses of hepatic artery thrombosis or biliary obstruction, despite the negative test for anti-HLA antibodies. The diagnosis of hyperacute rejection could be made associated with a short ischemic time and a good response after retransplantation.