Introduction: Fetal hydrocephalus, perinatal and neonatal represents a major therapeutic challenge and often has with poor neurological prognosis, due to its heterogeneous aetiology, including neurodevelopmental disorders.Objective: To make a collection of advances in cell therapy as a proposal that can extend the spectrum of treatment in congenita-neonatal hydrocephalus.Development: Therapeutic interventions available at present as the ventricle-peritoneal shunt and third ventriculostomy, are insufficient to fully resolve the hydrocephalus and to prevent or reverse the associated neurological damage.This is why what has emerged the need for new therapeutic alternatives based on the knowledge of physiopathological mechanisms involved in the development of this condition.In particular, cell therapy with neural stem cells and mesenchymal stem cells has proven in animal studies and preclinical studies with humans, efficiently and safely to prevent hydrocephalus originated from the disruption of the ventricular zone and secondary to intraventricular hemorrhage, with the consequent prevention of neurological sequelae sensorimotor and cognitive.Conclusions: So far, we do not have an efficient treatment that offers quality of life to patients with hydrocephalus, and that alternative therapy to be effective.