Hepatic graft-versus-host disease (GVHD) post allogeneic hematopoietic stem cell transplantation generally presents as cholestatic jaundice and increased serum alkaline phosphatase (ALK-P). Currently accepted standards for evaluating the clinical severity of hepatic GVHD are not based on serum aminotransferase levels but on the serum bilirubin levels. We describe a 25-year-old female who initially had no liver damage at all after an allogeneic peripheral blood stem cell transplantation (allo-PBSCT) from her HLA-indentical sister. Markedly elevated aminotransferases, without hyperbilirubinemia, however, developed 7 and 9 weeks after the first and second donor lymphocyte infusion (DLI), respectively. Liver biopsies performed in both events revealed lymphocytic infiltration of the portal tracts and pericentral necrosis of the lobuli. There was also a picture of periductal lymphocytic infiltration and vacuolization of the biliary epithelial cells, which was compatible with the diagnosis of GVHD of cholangiohepatitic type. These findings indicate that hepatic GVHD may present as acute hepatitis and should be included in the differential diagnosis for patients with increased aminotransferases after DLI.