In this study, we investigated the 3D chromatin organisation of the 11p15.5 imprinted region in cells from healthy individuals and from patients with BWS and SRS, and found that profound alterations in the chromatin architecture of the IGF2/H19 and CDKN1C/KCNQ1OT1 regions characterise both imprinting disorders. Here, KCNQ1OT1 is linked to Silver-Russell syndrome.