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II型糖原贮积病(酸性麦芽糖酶缺乏症)患者的基因缺陷

Genetic defects in patients with glycogenosis type II (acid maltase deficiency).

作者信息

Raben N, Nichols R C, Boerkoel C, Plotz P

机构信息

Arthritis and Rheumatism Branch, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD 20892, USA.

出版信息

Muscle Nerve Suppl. 1995;3:S70-4. doi: 10.1002/mus.880181415.

Abstract

Inherited deficiency of acid alpha-glucosidase (acid maltase, GAA) leads to glycogen storage disease type II. Clinical manifestations and prognosis of the disease depend on the age of onset and tissue involvement. GAA deficiency is extremely heterogeneous, ranging from a rapidly progressive fatal infantile-onset form to a slowly progressive adult-onset myopathy associated with respiratory insufficiency. Biochemical and immunochemical studies of the biosynthesis of the enzyme in GAA-deficient patients established the molecular diversity of the disease. Cloning and sequencing of the cDNA and the gene provided the basis for genetic analysis of the patients with different phenotypes. In this article, we summarize the data on mutations in the GAA gene and discuss the correlation between the genotype and phenotypic expression of the disease.

摘要

酸性α-葡萄糖苷酶(酸性麦芽糖酶,GAA)的遗传性缺乏会导致II型糖原贮积病。该疾病的临床表现和预后取决于发病年龄和组织受累情况。GAA缺乏具有极大的异质性,范围从快速进展的致命婴儿型到与呼吸功能不全相关的缓慢进展的成人型肌病。对GAA缺乏患者中该酶生物合成的生化和免疫化学研究确定了该疾病的分子多样性。cDNA和基因的克隆与测序为不同表型患者的遗传分析提供了基础。在本文中,我们总结了GAA基因突变的数据,并讨论了该疾病基因型与表型表达之间的相关性。

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