NeuroXsys will generate regulatory maps and models of the human X chromosome based on evolutionary conservation, with special attention to genes and regions implicated in X-linked neurological diseases. Vertebrate chromosomes are subdivided into domains of genomic regulatory blocks (GRBs) and NeuroXsys aims to map all GRBs on the X chromosome through bioinformatic approaches, extract gene regulatory sequences, and model their activity through transgenic reporter assays in the zebrafish juvenile and adult brain. One of the major deliverables of this project will be the publication of an online database that achieves correlation of disease genes ordered with respect to their regulatory regions and their experimentally and bioinformatically assessed function. NeuroXsys will seek to identify human disease mutations in neural gene regulatory elements. Implicated elements will be studied as regulators at single cell resolution in the zebrafish and mouse brain, defining expression patterns driven by the normal and mutant human regulatory DNA sequences. NeuroXsys will generate a regulatory map of a human chromosome.