阿尔及利亚脊髓肌肉萎缩症患者的临床和遗传学研究。

Journal of Neurodegenerative Diseases Pub Date : 2013-01-01 Epub Date: 2013-03-24 DOI:10.1155/2013/903875
Y Sifi, K Sifi, A Boulefkhad, N Abadi, Z Bouderda, R Cheriet, M Magen, J P Bonnefont, A Munnich, C Benlatreche, A Hamri
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引用次数: 0

摘要

脊髓性肌萎缩症(SMA)是第二种最常见的致死性常染色体隐性遗传疾病。它分为急性 Werdnig-Hoffmann 病(I 型)、中间型(II 型)、Kugelberg-Welander 病(III 型)和成人型(IV 型)。与所有四种形式的 SMA 相关的基因,即所谓的存活运动神经元(SMN)基因是重复的,有一个端粒拷贝(tel SMN 或 SMN1)和一个中心拷贝(cent SMN 或 SMN2)。在 95% 以上的 SMA 患者中,SMN1 被同源染色体删除。SMA的另一个候选基因是神经细胞凋亡抑制蛋白(NAIP)基因;在45-67%的I型和20-42%的II型/III型患者中,该基因出现同源缺失。在此,我们采用半定量 PCR 方法研究了来自 57 个无血缘关系家族的 92 名阿尔及利亚 SMA 患者(20 名 I 型、16 名 II 型、53 名 III 型和 3 名 IV 型)的 SMN 和 NAIP 基因。在75%的家族中发现了SMN1第7和/或第8外显子的同基因缺失。约 25% 的患者发现 NAIP 基因第 4 和/或第 5 外显子缺失。相反,SMN2拷贝的定量分析显示,SMN2拷贝数与SMA类型之间存在显著相关性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Clinical and Genetic Study of Algerian Patients with Spinal Muscular Atrophy.

Spinal muscular atrophy (SMA) is the second most common lethal autosomal recessive disorder. It is divided into the acute Werdnig-Hoffmann disease (type I), the intermediate form (type II), the Kugelberg-Welander disease (type III), and the adult form (type IV). The gene involved in all four forms of SMA, the so-called survival motor neuron (SMN) gene, is duplicated, with a telomeric (tel SMN or SMN1) and a centromeric copy (cent SMN or SMN2). SMN1 is homozygously deleted in over 95% of SMA patients. Another candidate gene in SMA is the neuronal apoptosis inhibitory protein (NAIP) gene; it shows homozygous deletions in 45-67% of type I and 20-42% of type II/type III patients. Here we studied the SMN and NAIP genes in 92 Algerian SMA patients (20 type I, 16 type II, 53 type III, and 3 type IV) from 57 unrelated families, using a semiquantitative PCR approach. Homozygous deletions of SMN1 exons 7 and/or 8 were found in 75% of the families. Deletions of exon 4 and/or 5 of the NAIP gene were found in around 25%. Conversely, the quantitative analysis of SMN2 copies showed a significant correlation between SMN2 copy number and the type of SMA.

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