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We performed this systematic review and meta-analysis to assess the efficacy and safety of anticoagulation therapy versus no anticoagulation in pediatric patients with cerebral sinovenous thrombosis (CSVT). Eligible studies included pediatric patients with CSVT that compared anticoagulation to no anticoagulation and reported on at least one relevant outcome (mortality, neurological deficit, thrombus resolution, recurrence, and bleeding). Meta-analysis reported risk ratios (RR) or differences (RD) (95% CIs) and absolute effects per 1000 patients. Risk of bias was assessed using ROBINS-I, and the certainty of evidence was evaluated with GRADE. After removing duplicates, we screened 8925 records independently and in duplicate, from which 12 nonrandomized studies of intervention were included. Compared with no anticoagulation, anticoagulation may be associated with lower risk of overall mortality RR 0.14 (0.05-0.39) (4 studies, n = 448), lower risk of neurological deficits RD -0.13 (-0.39 to 0.13) (6 studies, n = 462), and higher rates of complete and partial resolution RR 1.25 (0.94-1.66) (7 studies, n = 150). Recurrence RD 0 (-0.11 to 0.11) (2 studies, n = 56) and bleeding RD 0.03 (-0.09 to 0.15) (5 studies, n = 95) may be comparable between anticoagulation versus no anticoagulation. We judged outcomes to be very low to low certainty of evidence due to risk of bias, concerns from confounding, and imprecision from small sample sizes. To conclude, we show in this systematic review that anticoagulation therapy in pediatric patients with CSVT may be associated with better outcomes (reduced mortality, neurological deficits, and higher rate of thrombus resolution) with little to no effect on bleeding and recurrence.