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Cystic echinococcosis is a worldwide spread zoonosis and humans become accidental intermediate hosts. Any tissue can be affected. However, oromaxillofacial cystic echinococcosis is very uncommon and has never been studied systematically and no evidence-based treatment recommendations are available. Aim of this paper is to summarize the current knowledge and clinical experience with oromaxillofacial manifestations of cystic echinococcosis.PubMed database was accessed with a comprehensive, complex search strategy. Medical Subject Headings terms, wildcard search, truncated search terms and Boolean operators were used. No filters to restrict the results were set. Two evaluators jointly assessed the results in terms of defined criteria.The search strategy retrieved 538 results in total. After evaluation 83 publications presenting 108 cases on oromaxillofacial cystic echinococcosis were included. Parotid gland, mandibular region and maxillary region were most commonly affected. Median patient age was 22 years (range 2.5 months to 81 years). Surgical treatment was performed in 97 cases.In cases of oromaxillofacial cystic echinococcosis a thorough evaluation of the patients for further cysts is essential. Therapeutic treatment options are surgical or minimally invasive intervention and drug treatment with benzimidazoles. The diagnosis can be difficult and often require a multidisciplinary approach.