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ABSTRACT Background and Aims Primary sclerosing cholangitis (PSC) is an immune‐mediated cholestatic liver disease. Liver transplantation (LT) remains the only definitive treatment for patients with advanced disease. However, recurrence of PSC (rPSC) after LT is still a significant clinical challenge. The aim of this study was to assess the prevalence and incidence of rPSC following transplantation and to identify factors associated with its occurrence. Methods We conducted a systematic review and meta‐analysis of observational cohorts by searching PubMed, Embase and Cochrane for studies. Statistical analyses were conducted using R, applying a random‐effects model with 95% confidence intervals. Results Twenty‐nine studies were included, comprising 4682 patients who underwent LT for PSC (63.7% male). Most data originated from Europe (51.7%) and North America (29.2%), with additional cohorts from Asia (13.7%), South America (2.8%) and Oceania (2.6%). The worldwide pooled prevalence of rPSC was 18.66% (95% CI 15.33–22.52; I 2 = 84%) between 1980 and 2024. As for the worldwide pooled incidence, rPSC occurred at 26.04 per 1000 person‐years (95% CI 19.30–32.78; I 2 = 76.1%). The continent with the highest rPSC prevalence was Oceania with 30.65% (95% CI 23.17–39.29; I 2 = 13.8%) and South America had the highest incidence with 39.97 per 1000 person‐years (95% CI 25.42–54.51). Younger recipient age (mean difference MD = 5.16 years, 95% CI 4.13–6.19; I 2 = 51.3%), male sex (risk ratio RR = 0.92, 95% CI 0.87–0.98; I 2 = 0%), post‐LT cyclosporine use (RR = 0.56, 95% CI 0.42–0.76; I 2 = 0%), acute rejection (RR = 0.64, 95% CI 0.49–0.83) and de novo IBD (RR = 0.63, 95% CI 0.40–0.99; I 2 = 0%) were associated with an increased risk of recurrence. Conclusions Prevalence and incidence of rPSC post‐LT varied according to continents. Younger recipient age, male sex, de novo IBD, cyclosporine use post‐LT and acute rejection episodes were associated with a higher risk of recurrence. Trial Registration PROSPERO ID: CRD420250645485
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