Congenital anomalies of the kidneys and urinary tract (CAKUT) are a diverse group of structural malformations arising from disruption during embryonic development. While rare coding variants in developmental transcription factors, such as HNF1B, explain a subset of cases, most of these conditions remain genetically unexplained. We aim to characterise the molecular mechanisms underlying normal kidney and urinary tractdevelopment and understand what might be going wrong during these dynamic and cell-type specific programmes to result in structural malformations. To do this we combine clinical and whole genome sequencingdata from patients with multiomic maps of kidney development and functional genomic approaches to identify the gene-regulatory networks critical for kidney and urinary tract development and disease.