Dentigerous cyst is considered one of the representative cystic lesions, which accounts for approximately 15%-30% of the odontogenic cysts. Although its recurrence rate is low, a small proportion of dentigerous cysts converted into ameloblastomas, squamous cell carcinomas, and mucoepidermoid carcinomas. Here we present an uncommon case characterized by histopathological transformation from a dentigerous cyst to an ameloblastoma, and further investigate the factors contributing to its conversion.