Beutler E, Kuhl W
Proc Natl Acad Sci U S A. 1986 Oct;83(19):7472-4. doi: 10.1073/pnas.83.19.7472.
Fibroblasts from normal subjects and patients with the three types of Gaucher disease were labeled with [3H]leucine. Glucocerebrosidase antigen was immunoprecipitated using affinity-purified Sepharose-bound antibody. Normal cells initially formed a 60-kDa polypeptide antigen that was gradually replaced by a broad band of antigen averaging 63 kDa. This position corresponds with that of mature fibroblast and placental enzyme. Processing of glucocerebrosidase in six unrelated patients with type I Gaucher disease and one patient with type III Gaucher disease was exactly the same as normal. In contrast, three patients with the severe infantile (type II) form of the disease manifested a very unstable enzyme; the 60-kDa band appeared transiently and the mature 63-kDa band was never seen. These results indicate that type II Gaucher disease may well be distinguishable from type I disease by virtue of the very unstable enzyme precursor. Contrary to some earlier reports, processing of glucocerebrosidase in type I disease appears to be entirely normal.
用[3H]亮氨酸标记来自正常受试者以及三种戈谢病患者的成纤维细胞。使用亲和纯化的琼脂糖结合抗体对葡糖脑苷脂酶抗原进行免疫沉淀。正常细胞最初形成一种60 kDa的多肽抗原,该抗原逐渐被一条平均为63 kDa的宽条带抗原所取代。这个位置与成熟成纤维细胞和胎盘酶的位置相对应。6名无关的I型戈谢病患者和1名III型戈谢病患者的葡糖脑苷脂酶加工过程与正常情况完全相同。相比之下,3名患有严重婴儿型(II型)疾病的患者表现出一种非常不稳定的酶;60 kDa的条带短暂出现,从未见到成熟的63 kDa条带。这些结果表明,II型戈谢病很可能因其非常不稳定的酶前体而与I型疾病有所区别。与一些早期报告相反,I型疾病中葡糖脑苷脂酶的加工过程似乎完全正常。