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However, the onset and nature of kidney vascular abnormalities in ADPKD are poorly defined. Accordingly, we employed a combination of single-cell transcriptomics; three-dimensional imaging with geometric, topological and fractal analyses; and multimodal magnetic resonance imaging with arterial spin labelling to investigate aberrant microvasculature in ADPKD kidneys. Within human ADPKD kidneys with advanced cystic pathology and excretory failure, we identified a molecularly distinct blood microvascular subpopulation, characterised by impaired angiogenic signalling and metabolic dysfunction, differing from endothelial injury profiles observed in non-cystic human kidney diseases. Next, Pkd1 mutant mouse kidneys were examined postnatally, when cystic pathology is well established, but before excretory failure. An aberrant endothelial subpopulation was also detected, concurrent with reduced cortical blood perfusion. Disorganised kidney cortical microvasculature was also present in Pkd1 mutant mouse fetal kidneys when tubular dilation begins. Thus, aberrant features of cystic kidney vasculature are harmonised between human and mouse ADPKD, supporting early targeting of the vasculature as a strategy to ameliorate ADPKD progression.<\/jats:p>","DOI":"10.1242\/dmm.052024","type":"journal-article","created":{"date-parts":[[2025,3,21]],"date-time":"2025-03-21T08:05:55Z","timestamp":1742544355000},"source":"Crossref","is-referenced-by-count":8,"title":["Microvascular aberrations found in human polycystic kidneys are an early feature in a <i>Pkd1<\/i> mutant mouse model"],"prefix":"10.1242","volume":"18","author":[{"given":"Daniyal J.","family":"Jafree","sequence":"first","affiliation":[{"name":"UCL Great Ormond Street Institute of Child Health, 1 Developmental Biology and Cancer Research and Teaching Department , University College London, London WC1N 1EH ,","place":["UK"]},{"name":"UCL Centre for Kidney and Bladder Health, University College London 2 , London WC1E 6BT ,","place":["UK"]},{"name":"Specialised Foundation Programme 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