Abstract 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. Surveillance with CT imaging and clinical assessment at 6 months shows no evidence of recurrence.