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Introduction Keratocystic odontogenic tumors (KCOTs) are cystic tumours originating from the dental lamina of the maxilla and mandible that are lined with keratinized epithelium. While benign, they can be locally destructive and have a high recurrence rate despite treatment. Rarely, KCOTs may undergo malignant transformation into Primary Intraosseous Squamous Cell Carcinoma (PIOSCC). Case report This study reports the clinical findings, radiological scans and histopathology of 2 patients with KCOTs that underwent malignant transformation into PIOSCC. Discussion A comprehensive literature review was performed to similar reports documenting the malignant transformation of KCOTs. The potential for KCOTs to undergo malignant change should prompt oral maxillofacial surgeons and otolaryngologists to exercise a high index of suspicion when treating these lesions. Patients persisting with unresolved disease after treatment should be investigated for malignant transformation. Detailed histopathological examination of KCOT specimens is recommended to detect small foci of SCC which may be present in the epithelium.