The aim of this report is to present a rare case of a Ghost cell odontogenic carcinoma (GCOC) of a 21-year-old man with review of the literature. The patient was treated surgically, and one of the surgical margins was involved, the patient received adjuvant radiotherapy for local control. Five months later, patient presented with infraorbital lesion which was proven histological to be GCOC. Radiological images, histological sections and clinical photographs are also presented. One year after the second surgery, the patient was tumor free. The only effective treatment modality was surgical removal.