Chronic paroxysmal hemicrania (CPH) is a rare type of headache that is characterized by daily, multiple, short-lasting attacks of severe pain and associated autonomic symptoms. The pain is strictly unilateral and presents most commonly in the ocular, temporal, maxillary, and frontal areas. The excruciating, throbbing pain of CPH can be misdiagnosed as pain associated with dental pathology, especially when located in the maxillary area. Moreover, pain manifesting in the maxillary and temporal areas can be confused with temporomandibular disorders. CPH patients occasionally seek treatment in dental offices or orofacial pain centers. Accordingly, dentists should be familiar with CPH in order to avoid unnecessary, irreversible dental treatment. A case is presented to highlight many of the features of CPH.