Cholesteatoma of the paranasal sinuses is a rare pathology. A review of the literature reported less than 30 cases. These lesions mainly involve the frontal sinus.

A 25-year-old man presented with a right painful fronto-orbital mass associated with an ipsilateral eyelid oedema and a fever as high as 40°C. He experienced a general epileptic seizure requiring his admission in an intensive care unit.

CT-Scan with iodine injection evidenced the opacification of the right frontal sinus with bone lysis of the posterior wall. Complete surgical resection of a cystic structure containing keratin material was performed via eyebrow incision. The pathological examination confirmed the diagnosis of sinus cholesteatoma. Neurological signs entirely disappeared after surgery. Craniofacial MRI realized 2 months later showed no sign of recurrence. Obliteration of the right frontal sinus was performed 4 months later.

Although benign, cholesteatoma can spread to the surrounding structures leading to several complications including infections that can be life-threatening for the patient. CT-scan and MRI are useful examinations for diagnosis and follow-up. Complete surgical resection is required in order to avoid recurrence.