Department of Surgery, Washington University in St. Louis School of Medicine, St. Louis, Missouri, USA.
Department of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.
Am J Med Genet A. 2023 May;191(5):1339-1349. doi: 10.1002/ajmg.a.63181. Epub 2023 Mar 15.
The lack of United States population-based data on Turner syndrome limits assessments of prevalence and associated characteristics for this sex chromosome abnormality. Therefore, we collated 2000-2017 data from seven birth defects surveillance programs within the National Birth Defects Prevention Network. We estimated the prevalence of karyotype-confirmed Turner syndrome diagnosed within the first year of life. We also calculated the proportion of cases with commonly ascertained birth defects, assessed associations with maternal and infant characteristics using prevalence ratios (PR) with 95% confidence intervals (CI), and estimated survival probability. The prevalence of Turner syndrome of any pregnancy outcome was 3.2 per 10,000 female live births (95% CI = 3.0-3.3, program range: 1.0-10.4), and 1.9 for live birth and stillbirth (≥20 weeks gestation) cases (95% CI = 1.8-2.1, program range: 0.2-3.9). Prevalence was lowest among cases born to non-Hispanic Black women compared to non-Hispanic White women (PR = 0.5, 95% CI = 0.4-0.6). Coarctation of the aorta was the most common defect (11.6% of cases), and across the cohort, individuals without hypoplastic left heart had a five-year survival probability of 94.6%. The findings from this population-based study may inform surveillance practices, prenatal counseling, and diagnosis. We also identified racial and ethnic disparities in prevalence, an observation that warrants further investigation.
特纳综合征的美国人群数据缺乏,限制了对这种性染色体异常的患病率和相关特征的评估。因此,我们汇集了国家出生缺陷预防网络内的七个出生缺陷监测项目在 2000-2017 年的数据。我们估计了在生命的第一年被确诊为核型确诊特纳综合征的患病率。我们还计算了常规定性出生缺陷病例的比例,使用患病率比(PR)及其 95%置信区间(CI)评估了与母婴特征的关联,并估计了生存概率。任何妊娠结局的特纳综合征的患病率为每 10,000 名女性活产儿 3.2 例(95%CI=3.0-3.3,项目范围:1.0-10.4),活产儿和死产儿(≥20 周妊娠)的患病率为 1.9 例(95%CI=1.8-2.1,项目范围:0.2-3.9)。与非西班牙裔白人女性相比,非西班牙裔黑人女性出生的病例患病率最低(PR=0.5,95%CI=0.4-0.6)。主动脉缩窄是最常见的缺陷(占病例的 11.6%),在整个队列中,没有左心发育不良的个体五年生存率为 94.6%。这项基于人群的研究结果可能为监测实践、产前咨询和诊断提供信息。我们还发现了患病率方面的种族和民族差异,这一观察结果值得进一步调查。