A genetic model of neonatal hydrocephalus involving the coiled-coil domain-containing 39 (Ccdc39) gene may help elucidate cellular mechanisms leading to the abnormal accumulation of cerebrospinal fluid in neonatal hydrocephalus, as this model shows a robust and 100% penetrant hydrocephalus phenotype in mice, which is not common in other rodent models. Here, CCDC39 is linked to Hydrocephalus.