This type of neuropathic pain(atypical odontalgia) is seen most often in middle-aged women or men after dentoalveolar operation. Atypical odontalgia probably is caused by deafferentation leading to intraneural changes in the medullary dorsal horn. Treatment of this problem is difficult, but some success has been reported in uncontrolled, open-labeled studies using high doses of tricyclic antidepressants. This is the management report of a patient case, that had a neuropathic atypical odontalgia recognized with the right maxillary lateral incisor. The patient was consulted to the Department of Pain Clinics, ENT & Neurology and diagnosed the adenoid cystic carcinoma on left cerebellum and right paranasal sinus with extension to the cavernous sinus. In spite of the osteoplastic craniotomy, neurosurgical mass removal and radiation therapy were done with chemotherapy, atypical odontalgia was continued. In addition to the consultation to Psychology, stress management and antidepressant medication were done and improved slowly.