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人类红细胞丙酮酸激酶缺乏症:利用突变酶的动力学研究检测杂合子。

Human erythrocyte pyruvate kinase deficiency: the use of a kinetic study of mutant enzymes for the detection of heterozygotes.

作者信息

Sprengers E D, Marie J, Kahn A, Punt K, Staal G E

出版信息

Hum Genet. 1978 Feb 23;41(1):61-72. doi: 10.1007/BF00278872.

Abstract

Erythrocyte pyruvate kinase (PK) deficiency was detected in a boy of dutch origin. Immunologic, electrophoretic, and kinetic studies of the enzymes of propositus and the members of his family demonstrated that the boy was heterozygote for two different mutant PK alleles. The mutant enzyme, for which his mother was heterozygote, was characterized by a lower immunologic specific activity, a decreased affinity for the substrate phospho-enol-pyruvate, and a loss of homotropic interactions toward this substrate, an increased affinity toward the allosteric inhibitor MgATP2-, a decreased affinity for the activator fructose-1,6-diphosphate, and a lowered pH optimum. Electrophoresis, Km app. for MgADP, and the reactivity toward the purine nucleotide substrate analogues were normal. The mutant enzyme for which his father was heterozygote was characterized by a decreased affinity for the substrate phospho-enol-pyruvate and a loss of homotropic interactions toward this substrate. All other parameters mentioned above were normal. The use of a kinetic study of mutant enzymes for the detection of heterozygotes is discussed.

摘要

在一名荷兰裔男孩中检测到红细胞丙酮酸激酶(PK)缺乏症。对先证者及其家庭成员的酶进行免疫、电泳和动力学研究表明,该男孩是两种不同突变PK等位基因的杂合子。他母亲为杂合子的突变酶具有较低的免疫比活性、对底物磷酸烯醇丙酮酸的亲和力降低、对该底物的同促相互作用丧失、对变构抑制剂MgATP2-的亲和力增加、对激活剂果糖-1,6-二磷酸的亲和力降低以及最适pH值降低。电泳、MgADP的表观Km以及对嘌呤核苷酸底物类似物的反应性均正常。他父亲为杂合子的突变酶表现为对底物磷酸烯醇丙酮酸的亲和力降低以及对该底物的同促相互作用丧失。上述所有其他参数均正常。本文讨论了利用突变酶的动力学研究来检测杂合子的方法。

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