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脊髓性肌萎缩症的诊断

Spinal muscular atrophy diagnostics.

作者信息

Prior Thomas W

机构信息

Department of Pathology, Ohio State University, Columbus, Ohio 43210, USA.

出版信息

J Child Neurol. 2007 Aug;22(8):952-6. doi: 10.1177/0883073807305668.

Abstract

Spinal muscular atrophy is a common autosomal recessive neuromuscular disorder caused by mutations in the survival motor neuron gene (SMN), which exists in 2 nearly identical copies (SMN1 and SMN2). Exon 7 of SMN1 is homozygously absent in about 95% of spinal muscular atrophy patients, whereas the loss of SMN2 does not cause spinal muscular atrophy. Small mutations are found in the other 5% of affected patients, and these mutations cluster in the 3' end of SMN1, a region important for protein oligomerization. SMN1 dosage testing can be used to determine the SMN1 copy number and to detect spinal muscular atrophy carriers and affected compound heterozygotes. Dosage testing is compromised by the presence of 2 SMN1 copies per chromosome, which occurs in about 2% of carriers. Finally, although SMN2 produces less full-length transcript than SMN1, the number of SMN2 copies modulates the phenotype.

摘要

脊髓性肌萎缩症是一种常见的常染色体隐性神经肌肉疾病,由生存运动神经元基因(SMN)突变引起,该基因有两个几乎相同的拷贝(SMN1和SMN2)。在约95%的脊髓性肌萎缩症患者中,SMN1的外显子7纯合缺失,而SMN2的缺失不会导致脊髓性肌萎缩症。在另外5%的受影响患者中发现了小突变,这些突变聚集在SMN1的3'端,该区域对蛋白质寡聚化很重要。SMN1剂量检测可用于确定SMN1拷贝数,并检测脊髓性肌萎缩症携带者和受影响的复合杂合子。由于每条染色体存在2个SMN1拷贝,约2%的携带者会出现这种情况,这会影响剂量检测。最后,虽然SMN2产生的全长转录本比SMN1少,但SMN2拷贝数会调节表型。

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