Pseudotumor cerebri, also termed idiopathic intracranial hypertension, is defined by increased intracranial pressure in the setting of normal brain imaging and cerebrospinal fluid analysis. It can often be associated with optic nerve head edema. Several medications have been associated with pseudotumor cerebri,(1) including the use of lithium carbonate in the adult population(3) as well as in adolescents.(4) Signs and symptoms of increased intracranial pressure typically resolve after cessation of lithium carbonate usage.(5) We report a case of the onset of pseudotumor cerebri associated with lithium treatment in a child who sustained long-term optic atrophy and vision loss and required acetazolamide treatment for approximately 1 year after cessation of lithium.