Aim: There is scant evidence concerning the relationship of alarin concentrations for polycystic ovary syndrome (PCOS) status in the existing literature. Therefore, we aimed to reveal the relationship about predictive value of serum alarin concentrations for PCOS risk in infertile women. Methods: This prospective case-control study included a total of 151 infertile women who met eligibility criteria of the study. Infertile women diagnosed with PCOS formed the study group (n = 80). Women with diagnoses of unexplained infertility constituted the control group (n = 71). The biochemical analyses of serum concentrations of lipid profiles, estradiol (E2), follicle-stimulating hormone (FSH), luteinizing hormone (LH), anti-Mullerian hormone (AMH) and alarin were performed. Results: There were no differences for the study parameters, including age, body mass index, fasting glucose, total cholesterol, low-density lipoprotein cholesterol, high-density lipoprotein cholesterol, total triglyceride, E2, and FSH levels in either group. Serum LH, AMH, alarin concentrations, and antral follicle counts had higher values in the PCOS group compared with the controls. Correlation analysis revealed that serum alarin levels were significantly positively correlated with LH and AMH levels, only in the PCOS group. Multivariate binary logistic regression analysis demonstrated that infertile women with high alarin concentrations were significantly more likely to develop PCOS (OR = 1.77, 95% CI = 0.095-0.332, p < 0.001). Conclusion: Higher serum concentrations of alarin and a positive correlation with serum LH levels were found in infertile women with PCOS. This evidence supported that high alarin concentrations might play a role in the development of PCOS.