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一名生长发育迟缓婴儿中的三倍体嵌合体(45,X/68,XX)。

Triploidy mosaicism (45,X/68,XX) in an infant presenting with failure to thrive.

作者信息

Posey Jennifer E, Mohrbacher Nikki, Smith Janice L, Patel Ankita, Potocki Lorraine, Breman Amy M

机构信息

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas.

Texas Children's Hospital, Houston, Texas.

出版信息

Am J Med Genet A. 2016 Mar;170(3):694-8. doi: 10.1002/ajmg.a.37469. Epub 2015 Nov 14.

Abstract

Triploid mosaicism is a rare aneuploidy syndrome characterized by growth retardation, developmental delay, 3-4 syndactyly, microphthalmia, coloboma, cleft lip and/or palate, genitourinary anomalies, and facial or body asymmetry. In the present report, we describe a 3-month-old female presenting with failure to thrive, growth retardation, and developmental delay. A chromosomal microarray demonstrated monosomy X, but her atypical phenotype prompted further evaluation with a chromosome analysis, which demonstrated 45,X/68,XX mixoploidy. To our knowledge, this is the first report of a patient with this chromosome complement. Mosaicism in chromosomal aneuploidies is likely under-recognized and may obscure the clinical diagnosis. At a time when comparative genomic hybridization and genome sequencing are increasingly used as diagnostic tools, this report highlights the clinical utility of chromosome analysis when a molecular diagnosis is not consistent with the observed phenotype.

摘要

三倍体嵌合体是一种罕见的非整倍体综合征,其特征为生长发育迟缓、发育延迟、3-4指(趾)并指(趾)畸形、小眼畸形、脉络膜缺损、唇裂和/或腭裂、泌尿生殖系统异常以及面部或身体不对称。在本报告中,我们描述了一名3个月大的女性,表现为发育不良、生长发育迟缓及发育延迟。染色体微阵列显示X单体,但她的非典型表型促使进一步进行染色体分析,结果显示为45,X/68,XX混合倍体。据我们所知,这是首例具有这种染色体组成的患者报告。染色体非整倍体中的嵌合体可能未得到充分认识,并且可能会掩盖临床诊断。在比较基因组杂交和基因组测序越来越多地用作诊断工具的时代,本报告强调了当分子诊断与观察到的表型不一致时,染色体分析的临床实用性。

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