Objective To investigate the association between baseline serum Anti-Mullerian hormone (AMH) levels and IVF/ICSI outcomes in women with polycystic ovary syndrome (PCOS). Design Retrospective study. Setting Reproductive medicine center in a hospital. Population 2436 PCOS patients (Rotterdam criteria) who underwent their first fresh IVF/ICSI cycles were divided into three groups on the basis of the <25th (Group 1, n=611), 25 to 75th (Group 2, n=1216), or >75th (Group 3, n=609) percentile of baseline serum AMH level. Intervention(s) Baseline serum AMH levels measured on the 2-3 days of spontaneous menstrual cycle before IVF/ICSI treatment. Main Outcome Measure(s) Live birth rate (LBR), cumulative live birth rate (CLBR), clinical pregnancy rate (CPR), and normal fertilization rate (FR). Result(s) The LBR, CPR, and FR were significantly increased in Group 1 than Group 2 and Group 3, however, CLBR was similar between the three groups. The LBR were 46.6%, 40.5%, and 39.4% in Group 1, Group 2, and Group 3 respectively. The CPR were 53.0%, 47.0%, and 45.5%, respectively. The FR was highest in Group 1 (61.7%, P<0.05), but there was no uniform reverse trend with the AMH level. CLBR were 68.7%, 70.4%, and 71.3%, respectively. Although women in Group 1 were older (p < 0.05) and had higher body mass index (BMI) (p < 0.05), binomial logistic regression analysis used age, BMI, FSH, and AMH as independent variables indicated that only AMH was significantly associated with LBR and CPR. Nevertheless, binomial logistic regression analysis used age, BMI, FSH, AMH, and the number of retrieved oocytes as independent variables indicated that only the number of retrieved oocytes was significantly correlated with CLBR. After stratifying by age, the negative relationship between baseline AMH level and LBR and CPR remained only in the patients Conclusion(s) Higher baseline AMH level in PCOS women resulted in lower LBR, CPR, and FR but did not influence CLBR.