We report the case of a 24 year old man with a 22 year history of attacks of migraine without aura and attacks of ophthalmoplegic migraine. A persistent right 3rd nerve palsy appeared after the 17th attack. Although its existence has been debated, ophthalmoplegic migraine is recognized as such in the International Headache Society (IHS) classification. A review of published cases satisfying the IHS diagnostic criteria for ophthalmoplegic migraine is presented.