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人类神经氨酸酶缺乏症中的遗传异质性。

Genetic heterogeneity in human neuraminidase deficiency.

作者信息

Hoogeveen A T, Verheijen F W, d'Azzo A, Galjaard H

出版信息

Nature. 1980 Jun 12;285(5765):500-2. doi: 10.1038/285500a0.

Abstract

There is a deficiency of human alpha-N-acetylneuraminidase in several inherited diseases. In patients with mucolipidosis I (refs 1,2) and in adults with a variant form with out bony abnormalities and mental retardation, both also classified as sialidoses, it is the only deficient enzyme. In mucolipidosis II ('I-cell' disease) neuraminidase is one of many deficient lysosomal hydrolases and a third manifestation combines deficiency of neuraminidase and beta-galactosidase. We have investigated the genetic background of these various neuraminindase deficiencies by somatic cell hybridization and co-cultivation. The principal conclusions from work on mutant fibroblasts, reported here, are that at least three gene mutations are involved and that the combined beta-galactosidase/neuraminidase deficiency is likely to be due to defective post-translational modification of these enzymes.

摘要

在几种遗传性疾病中存在人α-N-乙酰神经氨酸酶缺乏的情况。在黏脂贮积症I患者(参考文献1,2)以及无骨骼异常和智力发育迟缓的变异型成年患者(这两种情况也归类为唾液酸沉积症)中,它是唯一缺乏的酶。在黏脂贮积症II(“I细胞”病)中,神经氨酸酶是多种缺乏的溶酶体水解酶之一,第三种表现形式是神经氨酸酶和β-半乳糖苷酶同时缺乏。我们通过体细胞杂交和共培养研究了这些不同的神经氨酸酶缺乏症的遗传背景。本文报道的关于突变成纤维细胞的研究主要结论是,至少涉及三个基因突变,并且β-半乳糖苷酶/神经氨酸酶联合缺乏可能是由于这些酶的翻译后修饰缺陷所致。

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