Abstract Background: Most tongue tumor lesions are evident upon physical examination; however, in cases where this does not happen in such a way, it is possible that invasion to adjacent neural structures may occur, resulting in headache as an initial manifestation. Case presentation: We present the case of a 57-year-old man with continuous hemicranial pain, in whom further studies revealed occult tongue carcinoma as the cause of the headache. After treatment and two-year observation, the patient has remained asymptomatic. Conclusions: This report suggests considering the tongue as a potential source of secondary etiologies of persistent hemicranial headache and describes a clinical variant of known cause of glossopharyngeal neuropathy (13.2.2.1 in ICHD III).