特纳综合征的心血管表现:核型亚型之间的表型差异

IF 1.5 4区 医学 Q3 CARDIAC & CARDIOVASCULAR SYSTEMS Pediatric Cardiology Pub Date : 2024-10-01 Epub Date: 2023-05-05 DOI:10.1007/s00246-023-03159-0
Alona Birjiniuk, Allison Goetsch Weisman, Christina Laternser, Joseph Camarda, Wendy J Brickman, Reema Habiby, Sheetal R Patel
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引用次数: 0

摘要

特纳综合征(TS)是一种遗传性疾病,表现为女性 X 染色体全部或部分单体。心血管异常很常见,包括先天性心脏缺陷(CHD)和主动脉扩张。虽然与非镶嵌型TS相比,镶嵌型TS的表型被怀疑不那么严重,但对不同核型之间心血管表现的差异还没有进行深入研究。这是一项单中心回顾性队列研究,包括2000年至2022年期间就诊的TS患者。研究回顾了人口统计学数据、染色体分析和影像学检查。核型分为单体X(45X)、45X嵌合、同染色质Xq、部分X缺失、环X(r(X))、带Y物质的TS及其他。采用皮尔逊卡方检验和韦尔奇双样本 t 检验比较了单体 X 和其他亚型的先天性心脏病和主动脉扩张患病率。我们共纳入了 182 名 TS 患者,中位年龄为 18 岁(4-33 岁)。与其他亚型相比,X-单体症患者中发生冠心病的比例更高(61.4% vs. 26.8%,P<0.05)。
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Cardiovascular Manifestations of Turner Syndrome: Phenotypic Differences Between Karyotype Subtypes.

Turner syndrome (TS) is a genetic disorder presenting in phenotypic females with total or partial monosomy of the X chromosome. Cardiovascular abnormalities are common, including congenital heart defects (CHD) and aortic dilation. Although mosaic TS is suspected to have less severe phenotype as compared to non-mosaic TS, differences in cardiovascular manifestations between karyotypes are not well studied. This is a single-center retrospective cohort study including patients with TS seen from 2000 to 2022. Demographic data, chromosomal analysis, and imaging were reviewed. Karyotypes were categorized as monosomy X (45X), 45X mosaicism, isochromosome Xq, partial X deletions, ring X (r(X)), TS with Y material, and others. Prevalence of CHD and aortic dilation were compared between monosomy X and other subtypes using Pearson's chi-square test and Welch two-sample t-test. We included 182 TS patients with median age 18 (range 4-33) years. CHD was more common in monosomy X as compared with others (61.4% vs. 26.8%, p < 0.001), including bicuspid aortic valve (44.3% vs. 16.1%, p < 0.001), partial anomalous pulmonary venous return (12.9% vs. 2.7%, p = 0.023), persistent left superior vena cava (12.9% vs. 1.8%, p = 0.008), and coarctation of the aorta (20.0% vs. 4.5%, p = 0.003). Cardiac surgery (24.3% vs. 8.9%, p = 0.017) was more prevalent in the monosomy X group. There was no statistically significant difference for presence of aortic dilation (7.1% vs 1.8%, p = 0.187). Although CHD and need for cardiac surgery are more common in TS with monosomy X as compared to others, all TS subtypes may have similar risk of developing aortic dilation. All TS patients should have similar cardiovascular surveillance testing to monitor for aortic dilation.

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来源期刊
Pediatric Cardiology
Pediatric Cardiology 医学-小儿科
CiteScore
3.30
自引率
6.20%
发文量
258
审稿时长
12 months
期刊介绍: The editor of Pediatric Cardiology welcomes original manuscripts concerning all aspects of heart disease in infants, children, and adolescents, including embryology and anatomy, physiology and pharmacology, biochemistry, pathology, genetics, radiology, clinical aspects, investigative cardiology, electrophysiology and echocardiography, and cardiac surgery. Articles which may include original articles, review articles, letters to the editor etc., must be written in English and must be submitted solely to Pediatric Cardiology.
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