II型糖原病(酸性麦芽糖酶缺乏症)患者的遗传缺陷。

N Raben, R C Nichols, C Boerkoel, P Plotz
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引用次数: 41

摘要

遗传性的α -葡萄糖苷酶(酸性麦芽糖酶,GAA)缺乏导致糖原储存病II型。该病的临床表现和预后取决于发病年龄和组织受累情况。GAA缺乏症是非常不均匀的,从快速进展的致命的婴儿发病形式到缓慢进展的成人发病肌病,并伴有呼吸功能不全。对gaa缺乏患者体内酶的生物合成进行生化和免疫化学研究,确定了该病的分子多样性。cDNA和该基因的克隆、测序为不同表型患者的遗传分析提供了依据。在本文中,我们总结了GAA基因突变的资料,并讨论了该疾病的基因型和表型表达之间的相关性。
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Genetic defects in patients with glycogenosis type II (acid maltase deficiency).

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.

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