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INTRODUCTION The aim of this paper is to present a rare case of frontal intersinus septal air cell inflammation as a cause of headaches. PRESENTATION OF CASE A 23-year-old patient was admitted to the Department of Otolaryngology and Laryngological Oncology for severe headaches during an upper respiratory tract infection. After neurological consultation including brain MRI and CT scanning of the paranasal sinuses, the inflammation of the frontal intersinus septal air cell was diagnosed. After examination, the patient was qualified for external osteoplasty. Under general endotracheal anaesthesia, the frontal intersinus septal air cell was intraoperatively opened from the side of the right frontal sinus, the mucoid content was aspirated and a plate of bone was removed. The patient reported complete relief from headaches on the second day after surgery. DISCUSSION According to the previous studies, the frontal intersinus septal cell is more frequent in patients with frontal sinus inflammation than in the patients without inflammatory changes. Further, the conducted research indicates that its occurrence does not result in significant disorders in the drainage and ventilation of the frontal sinuses, and as such is not likely to be the cause of inflammation. CONCLUSION In the case of inflammatory changes in the frontal intersinus septal air cell without concomitant frontal or ethmoid sinusitis, surgery via the external approach appears to be an effective method for the radical removal of pathological changes. [ABSTRACT FROM AUTHOR]