“…Anatomical characterization of these mice revealed severe congenital hydrocephalus that was nonetheless compatible with life and fertility in some cases. This obstructive hydrocephalus appeared to be secondary to failure of development of the subcommissural organ (SCO), a structure that is important for the patency of the aqueduct of Sylvius and normal cerebrospinal fluid flow in the brain (Cifuentes et al, 1994;Perez-Figares et al, 1998;PerezFigares et al, 2001;Rodriguez et al, 2001;Rodriguez et al, 1998;Vio et al, 2000). Identification of the genomic sequences flanking the inserted transgene led to the discovery that the transgene interfered with the expression of a novel brainspecific isoform of the winged helix transcription factor regulatory factor X4 (RFX4), which has been named RFX4 variant transcript 3, or RFX4_v3.…”