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一名患有18三体综合征的女性新生儿潜在22q11.2重复的检测

Detection of an Underlying 22q11.2 Duplication in a Female Neonate With Trisomy 18.

作者信息

Turbiville Donald E, Wu Hai, Dong Jianli

机构信息

School of Medicine.

Department of Pathology, University of Texas Medical Branch, Galveston, Texas.

出版信息

Lab Med. 2017 Nov 8;48(4):372-375. doi: 10.1093/labmed/lmx039.

Abstract

Current guidelines indicate that in patients with developmental disabilities or congenital anomalies, chromosomal microarray (CMA) is a first-tier diagnostic test. However, for patients with obvious chromosomal syndromes such as trisomy 13, 18, and 21, G-banded karyotyping is still recommended over CMA for establishing a diagnosis. In the case presented herein, a female neonate was suspected of having trisomy 18 based on pre- and postnatal evaluations. Karyotyping was requested but not performed due to insufficient cell growth; Interphase fluorescence in situ hybridization (i-FISH) found an extra copy of chromosome 18. CMA analysis uncovered gain of chromosome 18 and an additional duplication in chromosome 22q11.2, which went undetected with FISH. Our patient died within 40 hours after birth, but it is expected that patients with recognizable chromosomal syndromes could benefit from the discovery of coexisting copy number variations (CNVs) using CMA. This case shows that CMA can be a useful test for patients with recognizable chromosomal syndromes because of the potential benefits for patients and their families when co-existing CNVs are found.

摘要

当前指南指出,对于发育障碍或先天性异常患者,染色体微阵列分析(CMA)是一线诊断检测方法。然而,对于患有明显染色体综合征(如13三体、18三体和21三体)的患者,在进行诊断时,仍推荐采用G显带核型分析而非CMA。在本文所述病例中,一名女新生儿经产前和产后评估怀疑患有18三体。虽要求进行核型分析,但因细胞生长不足未进行;间期荧光原位杂交(i-FISH)发现有一条额外的18号染色体。CMA分析发现18号染色体增加以及22q11.2染色体存在额外重复,而FISH未检测到。我们的患者在出生后40小时内死亡,但预计患有可识别染色体综合征的患者可能会受益于通过CMA发现共存的拷贝数变异(CNV)。该病例表明,CMA对于患有可识别染色体综合征的患者可能是一种有用的检测方法,因为发现共存的CNV时可能会给患者及其家庭带来潜在益处。

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本文引用的文献

1
Anatomy of trisomy 18.18三体综合征的解剖学(情况)
Clin Anat. 2016 Jul;29(5):628-32. doi: 10.1002/ca.22725. Epub 2016 May 3.
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The trisomy 18 syndrome.18 三体综合征。
Orphanet J Rare Dis. 2012 Oct 23;7:81. doi: 10.1186/1750-1172-7-81.
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Clinical variability of the 22q11.2 duplication syndrome.22q11.2 重复综合征的临床变异性。
Eur J Med Genet. 2008 Nov-Dec;51(6):501-10. doi: 10.1016/j.ejmg.2008.07.005. Epub 2008 Jul 29.
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Familial 22q11.2 duplication: a three-generation family with a 3-Mb duplication and a familial 1.5-Mb duplication.
Clin Genet. 2008 Feb;73(2):160-4. doi: 10.1111/j.1399-0004.2007.00938.x. Epub 2007 Dec 12.

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