The keratocystic odontogenic tumor (KOT) is a lesion that requires special considerations because of its clinical behavior and its specific histopathological aspects. The present study aims to show a case of KOT with its radiographic and histopathological features. In this study a 43-year-old female patient presented with a unilocular radiolucent image located in the posterior region of the mandibular body on the left side, extending to the ascending ramus, were observed on a panoramic radiograph. Further, she undergoes a CT scan followed by biopsy for histopathological examination. The pathological cavity was lined by parkeratinized epithelium with a basal layer arranged in a “palisade” pattern cells with nuclear hyperchromatism were observed, confirming that it was a KOT. Therefore, dental surgeons’ knowledge of dental tumor of odontogenic tumor lesions, especially KOT is of fundamental importance to provide a correct diagnosis of these lesions, thereby preventing late diagnosis which can lead to the significant mutilation in the patients.
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