Background: Multiple sclerosis (MS) may have atypical presentations, one of whom being migraine-like headaches. This could be explained as following: the demyelination process in MS may involve brain structures or signaling pathways involved in migraine physiopathology, and therefore, simulate migraine headaches.
Case presentation: This case report is about a middle-aged male who developed a new-onset severe headache with migraine properties accompanied with vertigo, and was finally diagnosed with MS.
Conclusions: MS may have atypical presentations like severe headaches accompanied by additional symptoms, indicating the involvement of various brain structures; when working up such headaches, MS should be kept in mind besides other etiologies like vascular, infectious, and neoplastic conditions, and even primary migraine.