Hoepffner H J, Dreyer M, Reimers U, Schmidt-Preuss U, Koepp H P, Rüdiger H W
Arbeitsgruppe Toxikogenetik, Ordinariat für Arbeitsmedizin, Hamburg, Federal Republic of Germany.
Hum Genet. 1989 Oct;83(3):209-16. doi: 10.1007/BF00285157.
We describe a new familial syndrome in three siblings; it is biochemically characterized by a combined defect of the action of the three related peptides insulin, insulin-like growth factor I (IGF I) and epidermal growth factor (EGF). Clinically, the disease has features of Werner syndrome with lipodystrophy, scleroderma-like alterations of the skin, alterations of the skeleton and contractures of joints. In addition, one of the patients has an insulin-resistant diabetes mellitus. Studies with cultured fibroblasts obtained from skin biopsies show a markedly reduced stimulation of RNA synthesis by the three growth factors and a decreased insulin stimulation of 2-deoxy-D-glucose uptake as compared with normal controls. Receptor binding of the three peptides occurred with normal capacity and affinity. We conclude that the signal transfer of different growth factors has a common denominator at the postreceptor level.
我们描述了一种在三个兄弟姐妹中发现的新的家族综合征;其生化特征是胰岛素、胰岛素样生长因子I(IGF I)和表皮生长因子(EGF)这三种相关肽的作用存在联合缺陷。临床上,该疾病具有沃纳综合征的特征,包括脂肪营养不良、皮肤硬皮病样改变、骨骼改变和关节挛缩。此外,其中一名患者患有胰岛素抵抗型糖尿病。对取自皮肤活检的培养成纤维细胞的研究表明,与正常对照相比,这三种生长因子对RNA合成的刺激明显减少,胰岛素对2-脱氧-D-葡萄糖摄取的刺激也减少。这三种肽的受体结合能力和亲和力正常。我们得出结论,不同生长因子的信号转导在受体后水平有一个共同的特征。