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Background: The aim of the present systematic review was to summarize evidence on odontogenic carcinosarcoma, analyzing clinical, epidemiological, imaging, histopathological, immunohistochemical, therapeutic, and prognostic features of this tumor., Materials and Methods: This systematic review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Searches were performed in the Ovid MEDLINE (Wolters Kluwer), PubMed (National Library of Medicine), Web of Science (Thomson Reuters), Scopus (Elsevier), and LILACS (Latin American and Caribbean Center on Health Sciences Information) databases, without publication date or language restrictions. Case reports or case series of OCS reporting clinical, radiological, and histopathological data that confirmed the diagnosis were selected. The Joanna Briggs Institute-University of Adelaide tool was used for critical appraisal of the included articles., Results: Odontogenic carcinosarcoma is a rare, aggressive tumor associated with high mortality; however, the metastasis rate is low. The tumor has a male predilection. The mean patient age is 40 years, but there is no predilection for age. The left posterior mandible is the most affected site, but no specific radiographic features have been reported., Conclusion: Given its rarity, dentists, oral-maxillofacial surgeons, and physicians need to be aware of odontogenic carcinosarcoma in order to increase the diagnostic potential, preventing delays in diagnosis and treatment and thus contributing to lower morbidity of the tumor., (© 2024 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)