Pregnancy in chronic myeloid leukemia (CML) is uncommon. Management of leukemia during pregnancy and the effect of anticancer medication on pregnancy outcome is not well investigated. Imatinib, the target therapy for CML during pregnancy has been known to be associated with fetal teratogenicity, whereas withholding the treatment may leads to relapse of the disease. Here we successfully managed a mother with CML on imatinib, detected her pregnancy status incidentally in 2nd trimester, immediately stopped the medication. Subsequently a healthy baby was delivered with no apparent malformation.