Azathioprine vascular hepatotoxicity has been described mainly after renal transplantation. We report a new case in a patient who developed veno-occlusive disease of the liver 3 weeks after liver transplantation; regression of lesions were observed after discontinuation of azathioprine therapy. In this case, azathioprine hepatotoxicity may have been enhanced by a previous episode of severe, acute rejection prevailing in the hepatic veins. After liver transplantation, the diagnosis of azathioprine hepatotoxicity is difficult but should be suspected in the presence of non-inflammatory small hepatic veins lesions. Early withdrawal of the drug is mandatory for complete recovery.