Wagner D D, Mayadas T, Marder V J
J Cell Biol. 1986 Apr;102(4):1320-4. doi: 10.1083/jcb.102.4.1320.
Two conditions were identified that interfered with the complex polymerization process in biosynthesis of von Willebrand factor (vWf). Treatment of human umbilical vein endothelial cells with tunicamycin inhibited N-linked glycosylation of nascent vWf and the resulting pro-vWf monomers failed to dimerize. The single subunits accumulated in the endoplasmic reticulum and were neither processed further nor secreted. In the presence of a weak base (ammonium chloride or chloroquine), interdimer disulfide bond formation was inhibited in a dose-dependent manner. This process appeared therefore to be pH sensitive and likely to be initiated in the acidic trans-Golgi apparatus (Anderson, R. G. W., and R. K. Pathak, 1985, Cell, 40: 635-643). The weak base had no obvious effect on the other processing steps, i.e. dimerization, complex carbohydrate formation and sulfation, and produced only slight inhibition of prosequence cleavage. On the other hand, the weak base interfered with the targeting of newly synthesized vWf into Weibel-Palade bodies, with all of the vWf being secreted constitutively and none stored in the Weibel-Palade bodies. In summary, initial glycosylation of the nascent vWf protein and low pH in the trans-Golgi apparatus were important conditions for the successful polymerization of human vWf. Genetic defects disrupting any one of these conditions could result in the phenotype of von Willebrand disease.
已确定有两个条件会干扰血管性血友病因子(vWf)生物合成中的复杂聚合过程。用衣霉素处理人脐静脉内皮细胞会抑制新生vWf的N-连接糖基化,产生的前vWf单体无法二聚化。单个亚基在内质网中积累,既不进一步加工也不分泌。在弱碱(氯化铵或氯喹)存在的情况下,二聚体间二硫键的形成以剂量依赖的方式受到抑制。因此,这个过程似乎对pH敏感,可能在酸性反式高尔基体中启动(Anderson, R. G. W., and R. K. Pathak, 1985, Cell, 40: 635 - 643)。弱碱对其他加工步骤,即二聚化、复合碳水化合物形成和硫酸化没有明显影响,仅对前序列切割有轻微抑制。另一方面,弱碱干扰了新合成的vWf靶向魏尔-帕拉德小体,所有vWf都组成性分泌,没有一个储存在魏尔-帕拉德小体中。总之,新生vWf蛋白的初始糖基化和反式高尔基体中的低pH是人类vWf成功聚合的重要条件。破坏这些条件中任何一个的基因缺陷都可能导致血管性血友病的表型。