Atypical odontalgia is a pain disorder featured by constant pain without any obvious dental pathology. We report a 38-year-old man having chronic orofacial pain in the past two years. The pain was first located in the left maxillary premolar-molar region, but spread to the contra lateral mandible, occipital region, head and shoulders over time. The pain was described as dull and continuous with the intensity of #6 (VAS), and not relieved even after root canal therapy of all teeth and extraction of wisdom teeth . Some comorbid conditions such as TMJ problems and involvement of masticatory muscles were also reported. Finally, the diagnosis of atypical odontalgia was established due to lack of evidence for any organic pathology on clinical and paraclinical investigations, and a combination therapy including fluoxetine and clonazepam was started. Dental practitioners should consider AO when confronting toothache without any reasonable organic cause and avoid unnecessary dental procedures. KEYWORDS Atypical toothache; Malpractice; Endodontics.