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一个巴基斯坦家族中新发性少毛症位点的遗传定位到 7p21.3-p22.3 染色体及候选基因的筛选。

Genetic mapping of a novel hypotrichosis locus to chromosome 7p21.3-p22.3 in a Pakistani family and screening of the candidate genes.

机构信息

Department of Biochemistry, Faculty of Biological Sciences, Quaid-i-Azam University, Islamabad, Pakistan.

出版信息

Hum Genet. 2010 Aug;128(2):213-20. doi: 10.1007/s00439-010-0847-y. Epub 2010 Jun 11.

Abstract

Hereditary hypotrichosis is a heterogeneous group of inherited hair loss disorders characterized by diffused or localized thinning or absence of hair affecting scalp, eyebrows and eyelashes, and other body parts. Over the past few years, at least four autosomal dominant and six autosomal recessive forms of hypotrichosis have been described. All these ten forms of hypotrichosis have been mapped on different human chromosomes and the corresponding genes have been identified in most of these cases. In the present study, we have described a six-generation Pakistani consanguineous family with an autosomal recessive transmission of hereditary hypotrichosis. All the five affected individuals of the family showed complete absence of scalp hair and sparse eyebrows and eyelashes. They were born with complete absence of scalp hairs. Facial hair of beard and mustaches were present in all the affected adult male individuals. Papules were observed only on scalp of the affected individuals. A scalp biopsy from an affected individual showed markedly reduced number of hair follicles. Human genome scan using polymorphic microsatellite markers mapped the disease locus on chromosome 7p21.3-p22.3, flanked by markers D7S1532 and D7S3047. A maximum two-point LOD score of 4.74 (theta = 0.00) was obtained at marker D7S481. The linkage interval spans 15.69 cM, which corresponds to 6.59 Mb according to the sequence-based physical map (Build 36.2). Mutation analysis of five potential candidate genes (GNA12, FOXK1, DAGLB, ZNF12, ACTB), located in the linkage interval, did not reveal any functional sequence variant.

摘要

遗传性毛发稀少症是一组异质性遗传性脱发疾病,其特征为头皮、眉毛和睫毛以及其他身体部位的弥漫性或局限性变薄或缺失。在过去的几年中,已经描述了至少四种常染色体显性和六种常染色体隐性形式的毛发稀少症。所有这十种形式的毛发稀少症都定位于不同的人类染色体上,并且在大多数情况下已经确定了相应的基因。在本研究中,我们描述了一个六代巴基斯坦近亲家庭,其遗传性毛发稀少症呈常染色体隐性遗传。该家庭的所有 5 名受累个体均表现为头皮毛发完全缺失和稀疏的眉毛和睫毛。他们出生时就完全没有头皮毛发。所有受影响的成年男性个体均有胡须和髭毛。丘疹仅在受累个体的头皮上观察到。来自受累个体的头皮活检显示毛囊数量明显减少。使用多态性微卫星标记的人类基因组扫描将疾病基因座定位于 7p21.3-p22.3 染色体上,由标记 D7S1532 和 D7S3047 侧翼。在标记 D7S481 处获得最大两点 LOD 评分 4.74(theta = 0.00)。连锁间隔跨越 15.69 cM,根据基于序列的物理图谱(构建 36.2),相当于 6.59 Mb。位于连锁间隔内的五个潜在候选基因(GNA12、FOXK1、DAGLB、ZNF12、ACTB)的突变分析未发现任何功能序列变异。

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