Objective The study aims to investigate the ovarian reserve in premenopausal women with antiphospholipid syndrome (APS) and to evaluate whether it is associated with cumulative organ damage or the risk of clinical complications. Methods This single-center study was conducted in 23 premenopausal female patients (10 with primary APS and 13 with secondary APS) and 24 healthy volunteers. Serum anti-Müllerian hormone (AMH) levels were measured by enzyme-linked immunoassay. Disease-specific organ damage (DIAPS score) and the risk of clinical complications (aGAPSS score) were additionally evaluated in APS patients. Results Serum AMH levels were similar in APS patients (median 6.06, interquartile range 4.31–7.54 ng/ml) and in controls (4.87, 2.64–6.40 ng/ml; P = 0.116), and no differences were observed between the primary (6.60, 5.49–8.88 ng/ml) and secondary (6.06, 3.91–7.30 ng/ml; P = 0.532) forms of the syndrome. In individuals with APS, serum AMH levels correlated inversely with the aGAPSS score (rho–0.421, 95% confidence intervals −0.716 to −0.001; P = 0.045), while no associations were observed with the DIAPS score (rho–0.001, −0.423 to 0.422; P = 0.996). Conclusions Ovarian reserve is not reduced in premenopausal women with APS. In addition, serum AMH levels may reflect the risk of APS-related clinical complications but not the burden of disease-specific organ damage.