Although the cause of occipital neuralgia is mostly unknown, entrapment of the greater occipital nerve (GON) at its piercing point of the tendinous aponeurotic attachment of the trapezius at the superior nuchal line has been reported to be the most common cause. We report an occurrence of unilateral facial pain associated with continuous aching and stabbing headache in the frontotemporal and occipital regions in a patient whose occipital neuralgia has lasted for years. These symptoms were completely different from those of typical occipital neuralgia, making diagnosis very difficult. A 52-year-old male patient with a 6-year history of intermittent stabbing pain in his right suboccipital area presented with an unremitting continuous pain in his right frontotemporal and malar areas that lasted 5 months. The aching and stabling pain in his frontotemporal and malar areas developed suddenly 5 months prior to presentation. Although he was treated after visiting neurology, dentistry, opthalmology, ENT, and pain clinic, the pain was not controlled. It spread to right periorbital and right occipital areas. At 3 months following decompression of the right GON, he reported no more pain and was able to stop the medication. Until 12 months after the operation, his craniofacial pain did not recur.