Cerebral venous thrombosis (CVT) is a rare but life-threatening condition, particularly among pregnant and postpartum women. However, estimates of its incidence, recurrence, and associated adverse outcomes remain inconsistent. Moreover, the role of antithrombotic prophylaxis in mitigating these risks has not been fully established.This study aimed to synthesize global evidence on the incidence of CVT during pregnancy and postpartum, evaluate recurrence rates of CVT and noncerebral venous thromboembolism (VTE), assess adverse pregnancy outcomes, and explore the efficacy and safety of antithrombotic prophylaxis in high-risk populations.A systematic review and meta-analysis was conducted following PRISMA guidelines. Five major databases were searched for observational studies reporting CVT incidence, recurrence, or pregnancy outcomes in women with prior CVT or noncerebral VTE, covering the period from 1980 to September 2024, with an updated search completed on May 6, 2025. Data were pooled using random-effects models and heterogeneity was quantified via the I² statistic.Forty-seven studies encompassing 14,218 pregnancies were included. The pooled incidence of postpartum CVT was 7 cases per 100,000 deliveries (95% CI: 3-15), with significant regional disparities (Asia: 19/100,000; Europe: 3/100,000). The recurrence rate of CVT was 10.2 per 1,000 deliveries (95% CI: 5-21), while noncerebral VTE recurred in 15.3 per 1,000 pregnancies (95% CI: 8-28). The crude risk of spontaneous abortion was 15.7% (95% CI: 31.4-39.4), with higher rates observed in women not receiving prophylaxis. Antithrombotic prophylaxis appeared to reduce the risk of noncerebral VTE recurrence but did not significantly affect CVT recurrence.CVT and noncerebral VTE pose substantial risks during pregnancy, particularly in women with prior thrombotic events. Although antithrombotic prophylaxis may offer partial protection, its benefits must be carefully weighed against potential maternal and fetal risks. These findings underscore the need for individualized management and further research to inform evidence-based clinical guidelines.