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波兰脊髓性肌萎缩症家族中的连锁不平衡与单倍型分析。

Linkage disequilibrium and haplotype analysis among Polish families with spinal muscular atrophy.

作者信息

Brzustowicz L M, Wang C H, Matseoane D, Kleyn P W, Vitale E, Das K, Penchaszadeh G K, Munsat T L, Hausmanowa-Petrusewicz I, Gilliam T C

机构信息

Department of Psychiatry, Columbia University, New York.

出版信息

Am J Hum Genet. 1995 Jan;56(1):210-5.

Abstract

Spinal Muscular Atrophy (SMA) is an inherited degenerative disorder of anterior horn cells that results in progressive muscle weakness and atrophy. The autosomal recessive forms of childhood-onset SMA have been mapped to chromosome 5q11.2-13.3, in a number of studies examining different populations. A total of 9 simple sequence repeat markers were genotyped against 32 Polish families with SMA. The markers span an approximately 0.7 cM region defined by the SMA flanking markers D5S435 and MAP1B. Significant linkage disequilibrium (corrected P < .05) was detected at four of these markers, with D/Dmax values of < or = .89. Extended haplotype analysis revealed a predominant haplotype associated with SMA. The apparently high mutation rate of some of the markers has resulted in a number of haplotypes that vary slightly from this predominant haplotype. The predominant haplotype and these closely related patterns represent 25% of the disease chromosomes and none of the nontransmitted parental chromosomes. This predominant haplotype is present both in patients with acute (type I) and in chronic (types II and III) forms of SMA and occurs twice in a homozygous state, both times in children with chronic SMA.

摘要

脊髓性肌萎缩症(SMA)是一种遗传性的前角细胞退行性疾病,可导致进行性肌肉无力和萎缩。在多项针对不同人群的研究中,儿童期发病的常染色体隐性形式的SMA已被定位到5号染色体的q11.2 - 13.3区域。对32个患有SMA的波兰家庭的9个简单序列重复标记进行了基因分型。这些标记跨越了由SMA侧翼标记D5S435和MAP1B定义的约0.7厘摩区域。在其中四个标记处检测到显著的连锁不平衡(校正P <.05),D / Dmax值≤.89。扩展单倍型分析揭示了一种与SMA相关的主要单倍型。一些标记明显较高的突变率导致了许多与这种主要单倍型略有不同的单倍型。主要单倍型和这些密切相关的模式占疾病染色体的25%,在未传递的亲代染色体中均未出现。这种主要单倍型在急性(I型)和慢性(II型和III型)SMA患者中均存在,并且在纯合状态下出现过两次,均出现在慢性SMA儿童中。

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