Although it is currently unknown whether KIDINS220 variants in SINO and schizophrenic patients could lead to similar defects in SNX27-retromer and AQP4 expression and brain water dyshomeostasis as found in Kidins220 deficient mice, our data point at Kidins220f/f mice as an attractive model to study SINO syndrome, Kidins220-linked schizophrenia and other neuropathologies accompanied by ventriculomegaly such as foetal/perinatal to adulthood onset forms of hydrocephalus. Here, KIDINS220 is linked to schizophrenia.