We found that imprinted genes within the primary affected domains for SRS (IGF2 at 11p15.5) and PWS (SNURF and IPW at 15q11-q13) were down-regulated in all TS14 fibroblasts and in SRS/TS14 fibroblasts, despite the normal levels of methylation at their imprinted control regions, that are generally altered in SRS and PWS patients (6, 23). The gene discussed is IGF2; the disease is Prader-Willi syndrome.