Background: We present a case of paroxysmal tonic upgaze (PTU) of infancy treated with a daily low dose of Gravol ® to improve symptoms. Method: Case report Results: A one year-old boy presented with episodes of sustained conjugate upgaze that persisted for 30 to 45 minutes, varied in severity, and occurred with increasing frequency over the past two months. The episodes were worse when fatigued and were relieved by sleep. Pregnancy, delivery, and development were normal. Neurological examination between episodes was normal, as were EEG, brain MRI, and blood analysis. CSF neurotransmitter analysis showed serotonin and dopamine metabolites at lower levels of normal. The patient was diagnosed with paroxysmal tonic upgaze of infancy and was treated with 12.5 mg of Gravol ® daily with complete cessation of episodes. Conclusions: Paroxysmal tonic upgaze (PTU) of infancy is a disorder seen in infants where the eyes are forcibly deviated upwards for minutes to hours at a time. PTU often resolves spontaneously over several months, however episodes are extremely debilitating. Currently, treatments with levodopa have been tried with some success. Via its anticholinergic effects, Gravol may be a novel therapeutic option for PTU, negating the need to use serotonergic medications.