BACKGROUND : Cervicogenic headache (CEH) is characterized by unilateral posterior head and neck pain originating from cervical structures and may be improved or resolved by successful treatment of the causative cervical disorder or lesion. Cervical medial branch radiofrequency (RF) lesion therapy is effective in some CEH patients with no significant pathologic abnormalities that can be surgically corrected. However, patients with refractory CEH are often encountered clinically. METHODS : Here we describe the case of a 68-year-old female with an 18-year history of CEH who did not respond to a series of treatments including nonsteroidal antiinflammatory drugs, anticonvulsants, medial branch diagnostic block, medial branch RF lesion, and spinal cord stimulation. After careful examination, we performed percutaneous endoscopic right C2-C3 medial branch neurotomy under local anesthesia and neuroleptanalgesia. The patient's constant pain was ultimately relieved. CONCLUSIONS : For patients with refractory CEH who failed medial branch RF lesion, which could be relieved briefly by diagnostic medial branch injection, percutaneous endoscopic C2-C3 medial branches neurotomy may alleviate their pain under the premise of full informed consent, accurate localization, careful intraoperative exploration, and stimulation testing.