Ophthalmoplegia is a rare presentation of migraine. Ophthalmoplegic migraine is frequently encountered in children, but has rarely been observed in infancy. We report a case of an infant with paroxysmal episodes of vomiting followed by a few weeks long periods of oculomotor nerve palsy. The first episode occurred at 14 months of age and the second at two years. In both episodes full recovery of the ophthalmoplegia occurred without treatment. The differential diagnosis of painful ophthalmoplegia and the variety of therapeutic regimens are discussed.