Gemtuzumab ozogamicin (GO; CMA‐676; Mylotarg™) is a chemotherapeutic agent approved for the treatment of CD33‐positive acute myelogenous leukemia in patients of age 60 years or older after first relapse. Hepatic veno‐occlusive disease has been reported to develop as a late complication of gemtuzumab ozogamicin treatment. A patient who developed Budd‐Chiari Syndrome with hepatic vein thrombosis following treatment with GO is presented. This complication has not been previously reported, and it deserves to be considered as a possible adverse effect of gemtuzumab ozogamicin. Am. J. Hematol. 80:213–215, 2005. © 2005 Wiley‐Liss, Inc.