Beutler E, Kuhl W, Comings D
Am J Hum Genet. 1975 Sep;27(5):628-38.
The residual enzyme of the fibroblasts of a child with homozygous type 0 GM2 gangliosidosis (Sandhoff-Jatzkewitz disease) has been found to correspond with a minor fraction of enzyme which can be isolated from normal fibroblasts by repeated chromatography. This enzyme is designated as hexosaminidase (hex) S. It reacts with antiserum prepared against homogeneous hex A but not with serum prepared against homogeneous hex B. These findings support our previously described model of the relationship between hex A and hex G: hex A has the structure (alpha beta)3, while hex B is (beta)6. Type B GM2 gangliosidosis (Tay-Sachs disease) is the alpha- mutation, while type 0 GM2 gangliosidosis (Sandhoff-Jatzkewitz disease) is the beta- mutation. In the absence of normal beta subunits there is increased polymerization of alpha subunits forming hex S, which probably has a structure of (alpha)6. A parallel between the thalassemias and GM2 gangliosidosis is evident: deficiency of one of the chains of which the protein is composed leads to an excess of polymers comprised of the other chains. In type B GM2 gangliosidosis, the excess of beta chanis leads to increased amounts of hex B beta)6; in type 0 GM2 gangliosidosis, the excess of alpha chains leads to formation of increased amounts of the alpha chain polymer, hex S.
已发现一名纯合0型GM2神经节苷脂沉积症(桑德霍夫-雅茨凯维茨病)患儿成纤维细胞中的残留酶,与可通过反复层析从正常成纤维细胞中分离出的一小部分酶相对应。这种酶被命名为己糖胺酶(hex)S。它与针对纯合己糖胺酶A制备的抗血清发生反应,但不与针对纯合己糖胺酶B制备的血清发生反应。这些发现支持了我们之前描述的己糖胺酶A和己糖胺酶G之间关系的模型:己糖胺酶A具有(αβ)3结构,而己糖胺酶B是(β)6结构。B型GM2神经节苷脂沉积症(泰-萨克斯病)是α突变,而0型GM2神经节苷脂沉积症(桑德霍夫-雅茨凯维茨病)是β突变。在缺乏正常β亚基的情况下,α亚基的聚合增加,形成己糖胺酶S,其结构可能为(α)6。地中海贫血和GM2神经节苷脂沉积症之间的相似性很明显:蛋白质所由组成的其中一条链的缺乏会导致由其他链组成的聚合物过量。在B型GM2神经节苷脂沉积症中,β链过量导致己糖胺酶B(β)6的量增加;在0型GM2神经节苷脂沉积症中,α链过量导致α链聚合物己糖胺酶S的形成增加。