As endothelial re-expression of JAM-C failed to increase survival or inhibit hydrocephalus formation in JAM-C−/−//Tie2 JAM-C mice our observations point to non-endothelial JAM-C to be important for CNS homeostasis, although further investigations are required to elucidate its exact role in hydrocephalus pathogenesis. Here, TEK is linked to Hydrocephalus.