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5q11.2 缺失一例气管发育不良患者。

5q11.2 deletion in a patient with tracheal agenesis.

机构信息

Department of Clinical Genetics, Erasmus MC, Rotterdam, The Netherlands.

出版信息

Eur J Hum Genet. 2010 Nov;18(11):1265-8. doi: 10.1038/ejhg.2010.84. Epub 2010 Jun 16.

Abstract

Tracheal agenesis (TA) is a rare congenital anomaly of the respiratory tract. Many patients have associated anomalies, suggesting a syndromal phenotype. In a cohort of 12 patients, we aimed to detect copy number variations. In addition to routine cytogenetic analysis, we applied oligonucleotide array comparative genomic hybridization. Our patient cohort showed various copy number variations, of which many were parentally inherited variants. One patient had, in addition to an inherited 16p12.1 deletion, a 3.6 Mb deletion on chromosomal locus 5q11.2. This patient had a syndromic phenotype, including vertebral, anal, cardiovascular and tracheo-oesophageal associated anomalies, and other foregut-related anomalies, such as cartilage rings in the oesophagus and an aberrant right bronchus. No common deletions or duplications are found in our cohort, suggesting that TA is a genetically heterogeneous disorder.

摘要

气管发育不全(TA)是一种罕见的呼吸道先天性异常。许多患者存在相关畸形,提示存在综合征表型。在一组 12 名患者中,我们旨在检测拷贝数变异。除了常规细胞遗传学分析外,我们还应用了寡核苷酸微阵列比较基因组杂交。我们的患者队列显示了各种拷贝数变异,其中许多是父母遗传的变异。一名患者除了遗传的 16p12.1 缺失外,在染色体 5q11.2 位置还存在 3.6 Mb 的缺失。该患者具有综合征表型,包括椎体、肛门、心血管和气管食管相关畸形,以及其他前肠相关畸形,如食管软骨环和异常右支气管。我们的队列中未发现常见的缺失或重复,提示 TA 是一种遗传异质性疾病。

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