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一名黎巴嫩儿童患幼年型泰-萨克斯病的遗传病因。

Genetic cause of a juvenile form of Tay-Sachs disease in a Lebanese child.

作者信息

Boustany R M, Tanaka A, Nishimoto J, Suzuki K

机构信息

Duke University Medical Center, Durham, NC.

出版信息

Ann Neurol. 1991 Jan;29(1):104-7. doi: 10.1002/ana.410290120.

Abstract

Abnormality in the beta-hexosaminidase alpha gene underlying the clinical phenotype of a Lebanese patient with a juvenile form of Tay-Sachs disease has been studied. Clinical features were progressive spasticity, ataxia, and cognitive decline. The protein coding sequence of several beta-hexosaminidase alpha-chain complementary DNAs isolated by polymerase chain reaction was completely normal except for a G-to-A transition at nucleotide position 1511 within exon 13, which resulted in substitution of the normal arginine 504 (CGC) with histidine (CAC). Although the patient was from a first-cousin marriage, she was heterozygous for this mutation. The abnormality in the other allele, which is carried by the father, was not identified, except that it is neither of the two mutations responsible for the infantile Jewish Tay-Sachs disease. Biosynthetic and immunoprecipitation studies in cultured fibroblasts showed synthesis of the alpha-chain precursor, but the mature form of the alpha-subunit was not detected.

摘要

对一名患有青少年型泰-萨克斯病的黎巴嫩患者临床表型背后的β-己糖胺酶α基因异常进行了研究。临床特征为进行性痉挛、共济失调和认知衰退。通过聚合酶链反应分离出的几个β-己糖胺酶α链互补DNA的蛋白质编码序列完全正常,只是外显子13内核苷酸位置1511处发生了G到A的转换,导致正常的精氨酸504(CGC)被组氨酸(CAC)取代。尽管该患者来自近亲婚姻,但她对此突变是杂合子。未鉴定出父亲携带的另一个等位基因的异常情况,只是它不是导致婴儿型犹太泰-萨克斯病的两种突变之一。在培养的成纤维细胞中进行的生物合成和免疫沉淀研究表明,α链前体有合成,但未检测到α亚基的成熟形式。

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