The PDE1 family consists of three isoforms encoded by three distinct genes, PDE1A, PDE1B and PDE1C. We have shown that pde1a interference using splice- and translation-blocking morpholinos causes pronephric cysts, hydrocephalus, and body curvature in wild-type zebrafish embryos and aggravates the cystic phenotype in pkd2 morphants, while human PDE1A RNA partially rescues the pde1a and pkd2 morphant phenotypes [12]. This evidence concerns the gene PDE1A and Hydrocephalus.