To present a patient with a frontal sinus keratoma removed solely via endoscopic sinus surgery, including presentation of characteristic computed tomography and magnetic resonance images; to discuss the differential diagnosis of this condition; and to report the current knowledge on and treatment of frontal sinus keratoma.

A 53-year-old man presented to our department with a 10-month history of rhinorrhoea and postnasal drip. After computed tomography and magnetic resonance imaging studies, the patient underwent surgery utilising a modified Lothrop procedure. An extensive soft tissue lesion was removed from the frontal sinus. Histological examination revealed a lamellated cluster of keratinous material. The pathological diagnosis was keratoma of the frontal sinus. There was no recurrence of keratoma over a two-year follow-up period.

Following review of the English language literature, we believe this case report to represent the first successful application of a modified endoscopic Lothrop procedure for resection of an extensive frontal sinus keratoma. Thus, the applications of endoscopic sinus surgery may be expanded to include frontal sinus keratoma removal.