The aim of the systematic review is to analyze the type and the prevalence of dental side effects among cancer survivors treated with alkylating agents (AAs) during pediatric age. Moreover, the study aimed to investigate the association between the development of dental anomalies and the drug used or the tumor type. Four databases MEDLINE-PubMed, Web of Science, Scopus, and the Cochrane Central Register of Controlled Trials (CENTRAL) were searched from January 2024 to March 2024. All articles published up to March 2024 were evaluated. After removing duplicates, data extraction and risk of bias assessment using the Newcastle-Ottawa score were made. A summary of the overall strength of evidence available was performed using the "Grading of Recommendations Assessment, Development and Evaluation" (GRADE). Data were summarized using descriptive analysis as mean differences ± standard deviation or relative risks. Out of 2678 studies, the search identified five studies enrolled for the qualitative analysis of the data. Among 257 survivors, 155 (60.3%) reported: microdontia, agenesia, root shortening, enamel defects, and taurodontism. Microdontia occurred more frequently with other drugs compared to AAs. In conclusion, children treated with AAs showed microdontia (36.0%), root shortening (26.9%), and agenesis (23.5%). Secondly, the occurrence of dental anomalies was unaffected by drug treatment
thirdly, microdontia was the most frequent dental anomaly observed in both solid and lymphoproliferative tumors. This review was performed in accordance with the PRISMA guidelines. PROSPERO registration number CRD42023494560.