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三个患有异染性脑白质营养不良的乌克兰家庭中芳基硫酸酯酶A基因的新突变。

Novel mutations in arylsulfatase A gene in three Ukrainian families with metachromatic leukodystrophy.

作者信息

Olkhovich Natalia V, Takamura Noboru, Pichkur Natalia A, Gorovenko Natalia G, Aoyagi Kiyoshi, Yamashita Shunichi

机构信息

Department of Medical Genetics, Kyiv Medical Academy of Postdiploma Education, Kyiv, Ukraine.

出版信息

Mol Genet Metab. 2003 Nov;80(3):360-3. doi: 10.1016/j.ymgme.2003.08.004.

Abstract

Metachromatic leukodystrophy (MLD) is a lysosomal storage disease caused by the deficiency of arylsulfatase A (ARSA) or saposin B. The majority of mutations identified in patients with MLD are unique within individual families. Here, we report on the novel missense mutations (F247S, D381E, and A469G) and the known mutations "A" allele and P136S in the ARSA gene in three unrelated Ukrainian families with MLD. The mutations F247S and P136S were found in compound heterozygous with the "A" allele in two patients with juvenile onset MLD. The clinical features of the typical patient with genotype D381E/A469G (early onset with very rapid manifestation of disease) suggest the reason to distinguish an early infantile MLD variant.

摘要

异染性脑白质营养不良(MLD)是一种由芳基硫酸酯酶A(ARSA)或鞘脂激活蛋白B缺乏引起的溶酶体贮积病。在MLD患者中鉴定出的大多数突变在各个家庭中都是独特的。在此,我们报告了三个患有MLD的不相关乌克兰家庭中ARSA基因的新型错义突变(F247S、D381E和A469G)以及已知突变“A”等位基因和P136S。在两名青少年型MLD患者中发现F247S和P136S与“A”等位基因呈复合杂合状态。基因型为D381E/A469G的典型患者的临床特征(疾病早期发作且进展非常迅速)表明有理由区分出一种早期婴儿型MLD变体。

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