Plasma cell leukemia (PCL) is a rare variant of multiple myeloma, which is very aggressive and resistant to chemotherapy. We report a case of PCL successfully treated with syngeneic peripheral blood stem cell transplantation followed by low-dose thalidomide. As of March 2009, the patient has maintained CR for 39 months posttransplant. The clinical course of the present case suggests that autologous stem cell transplantation using a graft with reduced contamination of malignant cells followed by low-dose thalidomide maintenance therapy may improve the PCL treatment outcome.