Amenorrhea and galactorrhea developed in a female patient with tuberous sclerosis. There was no evidence of a pituitary tumor; she had an abnormal EEG, and computed tomographic scan showed multiple intracerebral calcifications but no lesions in the pituitary gland or hypothalamus. She had fixed hyperprolactinemia that was unresponsive to protirelin, chlorpromazine, levodopa, bromocriptine mesylate, or estrogen. The circulating prolactin may be of pituitary origin or may possibly be secreted ectopically by a hamartoma.