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Aim: The present systematic review and meta-analysis (Prospero registration number: CRD42023472016) aims to assess the prevalence of developmental defects of enamel (DDEs), qualitatively and/or quantitatively, in childhood cancer survivors (CCS) and evaluate, when possible, these data in comparison with those found in healthy children., Methods: Three electronic databases (PubMed, Embase, Scopus) were searched from January 2003 to January 2024 for studies reporting on DDEs in children with a mean age not exceeding 16 years at the time of the study who underwent antineoplastic therapy. The ROBINS-I and the Joanna Briggs Institute (JBI) tools were used to assess the risk of bias. Included studies with comparable outcomes underwent random effects models meta-analysis using Stata®18., Conclusion: CCS showed a higher prevalence of DDEs, both qualitative and quantitative, compared to healthy children. The meta- analysis showed higher odds of developing qualitative defects over quantitative defects in CCS. Conclusions regarding the association between the type of therapy administered, age of therapy initiation, and prevalence of DDEs could not be drawn due to insufficient data. A lack of a standardized method of detecting enamel defects posed a challenge in the qualitative and quantitative analysis.