Objective: Gremlin 1 and 2 regulate oocyte primordial follicle transition in animal models. The main objective of this study is to measure the blood levels of Gremlin 1 and 2 in the women with Polycystic Ovary Syndrome (PCOS). We also aimed to evaluate the association of these markers with hormonal and biochemical parameters of PCOS as interrupted folliculogenesis in those women is related to metabolic dysfunction.Material and methods: Fifty women with PCOS were diagnosed according to Rotterdam criteria, and thirty age-matched female controls were included in this prospective study. Gremlin 1 and 2 levels along with hormonal and metabolic parameters were compared between PCOS and control groups.Results: Serum Gremlin 1 levels were significantly higher in the PCOS group than in the control group (p=.001). Gremlin 2 levels were similar between the groups. Besides, there was a significant positive correlation between Gremlin 1 and insulin levels, Homeostasis Model Assessment-Insulin Resistance (HOMA-IR) and waist to hip ratio (WHR) (r=0.305; r=0.297; r=0.303, respectively).Conclusion: Our data suggest that Gremlin 1 may be the key regulator in the pathogenesis of PCOS. In future, Gremlin 1 may be a novel therapeutic target for the treatment of PCOS.