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10号染色体短臂上的缺失图谱绘制及第二个迪格奥尔格综合征基因座(DGS2)关键区域的定义。

Deletion mapping on chromosome 10p and definition of a critical region for the second DiGeorge syndrome locus (DGS2).

作者信息

Schuffenhauer S, Lichtner P, Peykar-Derakhshandeh P, Murken J, Haas O A, Back E, Wolff G, Zabel B, Barisic I, Rauch A, Borochowitz Z, Dallapiccola B, Ross M, Meitinger T

机构信息

Abteilung Medizinische Genetik, Ludwig-Maximilians-Universität München, Germany.

出版信息

Eur J Hum Genet. 1998 May-Jun;6(3):213-25. doi: 10.1038/sj.ejhg.5200183.

Abstract

DiGeorge syndrome (DGS) is a developmental field defect, characterised by absent/hypoplastic thymus and parathyroid, and conotruncal heart defects, with haploinsufficiency loci at 22q (DGS1) and 10p (DGS2). We performed fluorescence in situ hybridisations (FISH) and polymerase chain reaction (PCR) analyses in 12 patients with 10p deletions, nine of them with features of DGS, and in a familial translocation 10p;14q associated with midline defects. The critical DGS2 region is defined by two DGS patients, and maps within a 1 cM interval including D10S547 and D10S585. The other seven DGS patients are hemizygous for both loci. The breakpoint of the reciprocal translocation 10p;14q maps at a distance of at least 12 cM distal to the critical DGS2 region. Interstitial and terminal deletions described are in the range of 10-50 cM and enable the tentative mapping of loci for ptosis and hearing loss, features which are not part of the DGS clinical spectrum.

摘要

迪乔治综合征(DGS)是一种发育性场缺陷,其特征为胸腺和甲状旁腺缺如/发育不全以及圆锥动脉干心脏缺陷,22q(DGS1)和10p(DGS2)存在单倍剂量不足位点。我们对12例10p缺失患者(其中9例有DGS特征)以及1例与中线缺陷相关的家族性10p;14q易位进行了荧光原位杂交(FISH)和聚合酶链反应(PCR)分析。关键的DGS2区域由两名DGS患者确定,定位于一个1 cM区间内,包括D10S547和D10S585。其他7例DGS患者这两个位点均为半合子。相互易位10p;14q的断点位于关键DGS2区域远端至少12 cM处。所描述的中间缺失和末端缺失范围为10 - 50 cM,并初步确定了上睑下垂和听力丧失位点的图谱,这些特征不属于DGS临床谱。

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