无细胞DNA (cfNIPT)无创产前检测特纳综合征与镶嵌和结构变异-产前发现和产后结果。

IF 2.8 3区 医学 Q2 GENETICS & HEREDITY
Ivonne Bedei, Johanna Bruder, Ida C B Lund, Simon H Thomsen, Ida Vogel, Andrea T Maciel-Guerra, Francisco Alvarez-Nava, Melissa L Crenshaw, Roland Axt-Fliedner, Claus H Gravholt, Anne Skakkebæk
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引用次数: 0

摘要

特纳综合征(TS)是一种与核型异质性相关的性染色体疾病。虽然TS可能与严重的产前检查结果有关,最常与45x核型有关,但大多数TS胎儿没有明显的表型,导致诊断和治疗延迟。本研究的目的是评估无创产前无细胞DNA检测(cfNIPT)在检测具有不同TS核型变异的TS胎儿中的效果,并检查其表型变异和临床结果。从2000年到2024年,从德国胎儿超声专家那里收集了确诊或疑似TS的妊娠数据。此外,还包括少数丹麦病例,胎盘中有45,X嵌合现象。收集有关cfNIPT结果、核型、产前超声结果和妊娠结局的数据。114例患者中,100例(87.7%)为X单体cfNIPT高危结果,53例(46.5%)为真阳性(TP), 47例(41.2%)为假阳性(FP)。假阴性14例(12.3%)。TP和FN在先天性畸形和颈部半透明方面没有差异。对67例进行了核型分析。40例(59.7%)有45,X核型,16例(23.9%)有45,X嵌合,11例(16.4%)有结构变异。45x染色体核型与先天性畸形和颈部透明度增高相关(ps≤0.001)。与45,X染色体组相比,45,X染色体组的活产率更高(ps≤0.03)。出生后的表型通常是温和的。cfNIPT是早期识别TS核型变异胎儿的一种有价值的工具,能够及时干预和有针对性的管理。然而,高假阳性率强调了仔细咨询的必要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Non-Invasive Prenatal Testing by Cell-Free DNA (cfNIPT) for Detecting Turner Syndrome With Mosaicism and Structural Variants-Prenatal Findings and Postnatal Outcomes.

Turner Syndrome (TS) is a sex chromosomal disorder associated with karyotype heterogeneity. Although TS can be associated with severe prenatal findings, most often linked to the 45, X karyotype, the majority of TS fetuses have no overt phenotype, resulting in delayed diagnosis and management. The objective of this study is to assess the efficacy of non-invasive prenatal testing by cell-free DNA (cfNIPT) in detecting TS fetuses with different TS karyotype variants and to examine the phenotypic variations and clinical outcomes.Data on pregnancies with confirmed or suspected TS from 2000 to 2024 were collected from specialists in fetal ultrasound in Germany. In addition, a small number of Danish cases with 45, X mosaicism in the placenta was included. Data were collected regarding cfNIPT results, karyotypes, prenatal ultrasound findings, and pregnancy outcomes.Of the 114 cases included, 100 (87.7%) had a high-risk cfNIPT result for monosomy X, 53 (46.5%) were true positives (TP), and 47 (41.2%) were false positives (FP). Fourteen (12.3%) were false negatives (FN). No differences in congenital malformation or nuchal translucency were seen between TP and FN. Data on karyotype were available for 67 cases. Fourty (59.7%) had a 45, X karyotype, 16 (23.9%) 45, X mosaicism, and 11 (16.4%) had a structural variant. The 45, X karyotype was associated with a higher prevalence of congenital malformation and increased nuchal translucency (ps ≤ 0.001). The live birth rate was higher in cases with 45, X mosaicism or structural variants compared to cases with a 45, X karyotype (ps ≤ 0.03). Postnatal phenotypes were often mild.cfNIPT represents a valuable tool for the early identification of fetuses with TS karyotype variants, enabling timely intervention and targeted management. However, the high false-positive rate underscores the need for careful counseling.

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来源期刊
CiteScore
7.00
自引率
0.00%
发文量
42
审稿时长
>12 weeks
期刊介绍: Seminars in Medical Genetics, Part C of the American Journal of Medical Genetics (AJMG) , serves as both an educational resource and review forum, providing critical, in-depth retrospectives for students, practitioners, and associated professionals working in fields of human and medical genetics. Each issue is guest edited by a researcher in a featured area of genetics, offering a collection of thematic reviews from specialists around the world. Seminars in Medical Genetics publishes four times per year.
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