A 5-month-old boy and 7-month-old girl had episodes of downward eye deviation starting at age 5 months, lasting seconds to minutes, and associated with stiffening of the extremities in one case and grasping, flailing upper extremity movements and retroflexion of the head in the other. There were no other clinical abnormalities. Electroencephalography and MRI were normal. The episodes stopped after 6 to 12 weeks and there have been no sequelae. This idiopathic condition resembles paroxysmal tonic upgaze in infancy. Until further documentation clearly establishes that this phenomenon is benign, evaluation with MRI and electroencephalography is indicated.