Migraine is a common clinical diagnosis, occurring in 4-10% of school age children. Migraine in the infant and preschool child has been infrequently described in retrospective studies. We report the prospective evaluation and therapy of six children (5-42 months) with migraine. In four of the children, Prensky's criteria were used for diagnosis, while the two youngest children presented with ophthalmoplegic migraine. All children had a strong family history of migraine and presented with headache and prominent features including facial pallor, irritability, sleep disturbance or mood changes. The oldest four children were successfully treated with either amitriptyline or imipramine in low doses. The infants with ophthalmoplegic migraine failed to completely respond to any therapy. At followup 2 to 18 months later, all children were well and without toxicity. The pediatrician should be aware that migraine may begin in infancy and can be effectively and safely treated.