Introduction: Malignancy of dentigerous cyst into primary intraosseous carcinoma is infrequent and scantily documented in the literature. It represents 1% to 2.5% of the total odontogenic tumors and is exclusive of maxillary bones. It is more common among men aged around 50 years. Its clinical characteristics are edema, tooth mobility and paresthesia, and in most cases an absence of pain. These features hamper its diagnosis and pose a challenge to pathologists. Objective: Review the prevalence of primary intraosseous carcinoma derived from dentigerous cysts as published in PubMed in the last 15 years. Methods: A literature review was conducted of papers published in the last 15 years. The database PubMed was consulted using the following search terms: “dentigerous cyst prevalence”, “primary intraosseous squamous cell carcinoma”, “dentigerous primary intraosseous squamous cell carcinoma”. The papers included were in English or Spanish. Of the 217 papers obtained, 39 were selected for data crossing. Data analysis and integration: Recognition of a condition as aggressive as primary intraosseous carcinoma derived from a dentigerous cyst requires examination of its clinical and radiographic characteristics as well as its symptoms. Its close relationship to dentigerous cyst, the most prevalent of developmental cysts, makes it necessary to gain an accurate and deep understanding of both. Primary intraosseous carcinoma poses a challenge to clinicians due to its low symptomatology and poor survival. Of the total 44 823 odontogenic cysts studied, 9 806 were diagnosed as dentigerous cysts and 22 maligned into primary intraosseous carcinoma, for 0.32% of the total. Conclusions: Malignation of a dentigerous cyst into primary intraosseous carcinoma has a low incidence and a silent presentation, which hampers its early diagnosis. Protocol biopsy is recommended for each case of dentigerous cyst to prevent the poor survival caused by primary intraosseous carcinoma.