Two children with neurofibromatosis and a chief complaint of severe, episodic, unilateral facial itching were found to have brainstem glioma. Initial computerized tomography of the brain was thought to be normal, but the brainstem tumor was subsequently demonstrated on magnetic resonance imaging. The paroxysmal facial itching resolved in both cases after a course of radiation therapy. The pathophysiology of the attacks of facial itching is unknown but must be related to the brainstem glioma. This is the first report of paroxysmal, unilateral facial itching as a presenting sign of childhood brainstem tumor. (J Child Neurol 1988;3:189-192).