A paranasal sinus disease complicated with headache may be misdiagnosed as a neurological disease, which may result in delay of appropriate management on the underlying sinus disease. Not only inflame-matory but also non-inflammatory diseases of the paranasal sinuse, e. g. the submu-cosal hematoma ov frontal sinus, can the pressure change and irritation of the sinus mucosa. We reported a case of submucosal hematoma of the frontal sinus with initial symptom of headache. Through and osteoplastic approach, the hematoma was successfully removed and proven histo-logically. Besides, the mechanism of paranasal pain caused by the frontal sinus hematoma was discussed.