Thirteen neurologically impaired children with paroxysmal ocular downward deviation were studied. The eye movement accompanied downward movement of the upper eyelid; there was no coincident paroxysmal discharge on electroencephalography. The ocular abnormality was observed in infancy, predominantly in preterm infants, and spontaneously resolved in many of them. All patients had spastic quadriplegia or diplegia and mental retardation. Many children also had cortical visual impairment. This eye movement abnormality, not reported previously, should be considered to be a neurologic sign in brain-damaged infants with cortical visual impairment.