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CONTEXT Antimüllerian hormone (AMH) levels are higher in patients with polycystic ovary syndrome (PCOS). Accumulating evidence indicates that AMH has an impact on the physiology of the female reproductive system. OBJECTIVE To investigate the association of AMH levels with the risk of preterm delivery in PCOS patients. DESIGN Retrospective cohort study. SETTING Academic fertility center. PATIENTS Women who underwent in vitro fertilization between January 2017 and July 2018 (25,165 cycles). INTERVENTIONS None. MAIN OUTCOME MEASURES The primary outcome was preterm delivery. RESULTS Serum AMH levels were not different between the term delivery and preterm delivery groups in the entire cohort (3.8 vs. 4.1 ng/mL, P>.05). In patients diagnosed with PCOS, those with preterm delivery had higher AMH levels than were found in patients with term delivery (9.3 vs. 6.9 ng/mL, P<.01). Preterm deliveries predominated in PCOS patients with AMH levels above the 75th percentile (9.75 ng/mL) (adjusted P<.0001, adjusted odds ratio [OR] 4.0, 95% confidence interval [CI] 1.94, 8.08)) and frozen-thawed embryo transfer (FET) patients with AMH levels higher than the 90th percentile (10.10 ng/mL) (adjusted P<.05, adjusted OR 2.0, 95% CI 1.16, 3.36). CONCLUSION Serum AMH levels higher than 75th percentile were associated with an increased risk of preterm delivery in patients with PCOS, and serum AMH levels higher than the 90th percentile were associated with an increased risk of preterm delivery in FET patients.