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3q29微缺失综合征:一种新综合征的临床与分子特征

3q29 microdeletion syndrome: clinical and molecular characterization of a new syndrome.

作者信息

Willatt Lionel, Cox James, Barber John, Cabanas Elisabet Dachs, Collins Amanda, Donnai Dian, FitzPatrick David R, Maher Eddy, Martin Howard, Parnau Josep, Pindar Lesley, Ramsay Jacqueline, Shaw-Smith Charles, Sistermans Erik A, Tettenborn Michael, Trump Dorothy, de Vries Bert B A, Walker Kate, Raymond F Lucy

机构信息

Department of Medical Genetics, Addenbrooke's Hospital, University of Cambridge, Cambridge, United Kingdom.

出版信息

Am J Hum Genet. 2005 Jul;77(1):154-60. doi: 10.1086/431653. Epub 2005 May 25.

DOI:10.1086/431653
PMID:15918153
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC1226188/
Abstract

We report the identification of six patients with 3q29 microdeletion syndrome. The clinical phenotype is variable despite an almost identical deletion size. The phenotype includes mild-to-moderate mental retardation, with only slightly dysmorphic facial features that are similar in most patients: a long and narrow face, short philtrum, and high nasal bridge. Autism, gait ataxia, chest-wall deformity, and long and tapering fingers were noted in at least two of six patients. Additional features--including microcephaly, cleft lip and palate, horseshoe kidney and hypospadias, ligamentous laxity, recurrent middle ear infections, and abnormal pigmentation--were observed, but each feature was only found once, in a single patient. The microdeletion is approximately 1.5 Mb in length, with molecular boundaries mapping within the same or adjacent bacterial artificial chromosome (BAC) clones at either end of the deletion in all patients. The deletion encompasses 22 genes, including PAK2 and DLG1, which are autosomal homologues of two known X-linked mental retardation genes, PAK3 and DLG3. The presence of two nearly identical low-copy repeat sequences in BAC clones on each side of the deletion breakpoint suggests that nonallelic homologous recombination is the likely mechanism of disease causation in this syndrome.

摘要

我们报告了6例3q29微缺失综合征患者的鉴定情况。尽管缺失大小几乎相同,但临床表型存在差异。表型包括轻度至中度智力障碍,多数患者仅有轻微的面部畸形特征:脸长且窄、人中短、鼻梁高。6例患者中至少有2例出现自闭症、步态共济失调、胸壁畸形以及手指细长。还观察到其他特征——包括小头畸形、唇腭裂、马蹄肾和尿道下裂、韧带松弛、复发性中耳感染以及色素沉着异常——但每个特征仅在1例患者中出现过一次。该微缺失长度约为1.5 Mb,在所有患者中,分子边界均定位于缺失两端相同或相邻的细菌人工染色体(BAC)克隆内。该缺失包含22个基因,包括PAK2和DLG1,它们分别是两个已知X连锁智力障碍基因PAK3和DLG3的常染色体同源基因。缺失断点两侧的BAC克隆中存在两个几乎相同的低拷贝重复序列,这表明非等位基因同源重组可能是该综合征致病的机制。

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Screening for subtelomeric rearrangements in 210 patients with unexplained mental retardation using multiplex ligation dependent probe amplification (MLPA).使用多重连接依赖探针扩增技术(MLPA)对210例不明原因智力障碍患者进行亚端粒重排筛查。
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