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The Odontogenic Keratocyst (OKC) is one of the most aggressive odontogenic cysts. OKCs of the maxilla are particularly rare with less than 1% of cases reported in the literature. A 29-year-old female patient presented with pain and loose upper molars. Imaging confirmed an ectopic tooth at the osteomeatal complex and a maxillary OKC. These were endoscopically surgically removed and two teeth were encountered at the maxillary antrum. Histopathology confirmed the diagnosis of OKC of the maxilla.doi:10.1093/jscr/rjac078 pmid:35422993 pmcid:PMC9004408 fatcat:67taduryfrbarkq3vv5uk7dizm