OBJECTIVE: To review the entity of respiratory epithelial adenomatoid hamartoma of the paranasal sinuses using an illustrative case example of frontal sinus hamartoma. METHODS: Case report with review of the literature. RESULTS: Respiratory epithelial adenomatoid hamartoma represents a benign process that is generally cured with conservative surgical resection of the involved area. Nevertheless, as shown in our case example, this can be a locally aggressive process that can lead to significant morbidity if left untreated. CONCLUSION: Hamartomas of the paranasal sinuses represent a rare entity, which should be considered in the differential diagnosis of expansile sinonasal lesions. Treatment of choice is surgical resection. Frontal sinus involvement may extend into the surrounding orbit and intracranial cavities, which may need to be addressed with standard skull base surgery techniques.