CFHR1 and hemolytic-uremic syndrome: Here, we describe two patients with HUS who underwent a KT from an uncontrolled DCD (uDCD).<h4>Case summary</h4>The first patient, affected by aHUS due to a heterozygous deletion in CFHR3-CFHR1 and a novel heterozygous variant in CFHR5 gene, underwent a KT with eculizumab prophylaxis.