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由于 16q24.1 缺失导致肺静脉错位的肺泡毛细血管发育不良的早期产前诊断。

Early prenatal diagnosis of alveolar capillary dysplasia with misalignment of pulmonary veins due to a 16q24.1 deletion.

机构信息

Laboratoire de Cytogénétique, Service d'Histologie-Embryologie-Cytogénétique, Hôpital Antoine Béclère, AP-HP Université Paris Saclay, Clamart, France.

UF de Foetopathologie, Hôpital Antoine Béclère, AP-HP Université Paris Saclay, Clamart, France.

出版信息

Am J Med Genet A. 2021 May;185(5):1494-1497. doi: 10.1002/ajmg.a.62105. Epub 2021 Jan 31.

Abstract

First trimester ultrasound screening is an essential fetal examination performed generally at 11-13 weeks of gestation (WG). However, it does not allow for an accurate description of all fetal organs, partly due to their development in progress. Meanwhile, increased nuchal translucency (INT) is a widely used marker known to be associated with chromosomal deleterious rearrangements. We report on a 14 WG fetus with an association of INT and univentricular congenital heart malformation (CHM) leading to chorionic villous sampling (CVS). Cytogenetic investigations performed using array-Comparative Genomic Hybridization (CGH) and fluorescence in situ hybridization (FISH) demonstrated a 1.17 Mb deletion in 16q24.1 encompassing FOXF1 arisen de novo on maternal inherited chromosome. Fetopathological study confirmed CHM with hypoplastic left heart syndrome (HLHS) associating aortic atresia, mitral stenosis, and left ventricular hypoplasia and revealed in addition specific lung lesions corresponding to alveolar capillary dysplasia with misalignment of pulmonary veins (ACDMPV). This is so far the first case of first trimester prenatal diagnosis of ACDMPV due to the deletion of FOXF1 gene. An interpretation of the complex genomic data generated by ultrasound markers is facilitated considerably by the genotype-phenotype correlations on fetopathological examination.

摘要

早孕期超声筛查是一项重要的胎儿检查,通常在妊娠 11-13 周进行。然而,它不能准确描述所有胎儿器官,部分原因是它们正在发育中。同时,颈项透明层(NT)增厚是一种广泛使用的标志物,已知与染色体有害重排有关。我们报告了一例 14 周妊娠胎儿,其颈项透明层增厚与单心室先天性心脏畸形(CHM)相关,导致绒毛膜取样(CVS)。使用阵列比较基因组杂交(CGH)和荧光原位杂交(FISH)进行的细胞遗传学研究显示,FOXF1 基因在母系遗传染色体上发生了 1.17Mb 的缺失。胎儿病理学研究证实 CHM 伴有左心发育不全综合征(HLHS),伴主动脉瓣闭锁、二尖瓣狭窄和左心室发育不良,并发现了与肺静脉排列不齐相关的肺泡毛细血管发育不良(ACDMPV)的特定肺部病变。这是迄今为止由于 FOXF1 基因缺失而导致的早孕期产前诊断 ACDMPV 的首例病例。通过对胎儿病理学检查的基因型-表型相关性分析,可以极大地解释由超声标志物产生的复杂基因组数据。

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