Cerebral ventriculomegaly and hydrocephalus are not frequently associated with endocrine disorders of the gonadotropic axis. The mechanism of this association is not clarified. The most probable cause is however a partial hypothalamic dysfunction. The examination of the few reported cases is in favour of this explanation. We present the case of a young woman with a cerebral ventriculomegaly and suffering from secondary amenorrhea. Shunt was not necessary from the neurological point of view, the problem of secondary amenorrhea and anovulatory infertility was solved by clomiphen citrate therapy.