ABSTRACT Dentigerous cysts frequently develop as odontogenic cysts in the oral cavity that can occasionally progress to neoplasms. Tumor from dentigerous cysts is uncommon, with only 42 occurrences documented in the literature. This case report describes a 73-year-old male who developed primary intraosseous squamous cell carcinoma after a dentigerous cyst. The dental practitioner should have knowledge of the potential for malignant transformation in asymptomatic dentigerous cysts and ensure thorough follow-up. Even if the lesion appears benign, it is recommended to perform a complete histological investigation. Histopathological investigations have revealed malignancies in the follicle that surrounds the dental crown. While malignant lesion formation is uncommon, it should be considered a differential diagnosis in appropriate circumstances.