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兄弟姐妹中先天性心脏缺陷的复发——一项全国性研究。

Recurrence of congenital heart defects among siblings-a nationwide study.

作者信息

Brodwall Kristoffer, Greve Gottfried, Leirgul Elisabeth, Tell Grethe S, Vollset Stein E, Øyen Nina

机构信息

Department of Global Public Health and Primary Care, University of Bergen, Bergen, Norway.

Department of Pediatrics, Haukeland University Hospital, Bergen, Norway.

出版信息

Am J Med Genet A. 2017 Jun;173(6):1575-1585. doi: 10.1002/ajmg.a.38237. Epub 2017 Apr 19.

Abstract

Congenital heart defects (CHD) constitute the largest group of congenital malformations. In most families, only one person has CHD; however, the risk of CHD increases for children born into families already affected. In this study, all births from 1994 through 2009 were identified in the Medical Birth Registry of Norway, including supplemental information on CHD from clinical and administrative registers, as part of the CVDNOR project. By using the unique personal identification number of each parent we were able to link 16,078 pairs of twins, 445,584 pairs of full siblings, and 106,840 pairs of half-siblings. Sibling recurrence risk ratio (RRR) was calculated using CHD status in the oldest sibling as exposure and CHD status in the younger sibling as outcome, adjusted for year of birth, maternal age, and maternal diabetes. Among full sibling pairs with CHD in the older sibling, the younger sibling had CHD in 4.1% compared to 1.1% of the pairs without CHD in the older sibling (adjusted RRR 3.6; 95% confidence interval (CI) 3.1-4.1). In same-sex twins the RRR was 14.0 (95% CI 10.6-18.6), and in opposite-sex twins the RRR was 11.9 (95% CI 7.1-19.9). For half-siblings the RRR was 1.5 (95% CI 0.8-2.8). When restricting to severe types of CHD, the RRR was 6.9 (95% CI 4.9-9.8) for full siblings. In 50% of the pairs with recurrent CHD, the siblings had similar types of CHD. The high relative risk of recurrence indicates that familial risk factors are important in the etiology of CHD.

摘要

先天性心脏病(CHD)是先天性畸形中最大的一组。在大多数家庭中,只有一人患有先天性心脏病;然而,对于出生在已有患者的家庭中的儿童,患先天性心脏病的风险会增加。在这项作为CVDNOR项目一部分的研究中,通过挪威医疗出生登记处识别了1994年至2009年的所有出生情况,包括来自临床和行政登记处的关于先天性心脏病的补充信息。通过使用每位父母唯一的个人识别号码,我们能够将16,078对双胞胎、445,584对全同胞和106,840对半同胞进行关联。以年长同胞的先天性心脏病状况为暴露因素,以年幼同胞的先天性心脏病状况为结果,计算同胞复发风险比(RRR),并对出生年份、母亲年龄和母亲糖尿病进行调整。在年长同胞患有先天性心脏病的全同胞对中,年幼同胞患先天性心脏病的比例为4.1%,而在年长同胞未患先天性心脏病的同胞对中这一比例为1.1%(调整后的RRR为3.6;95%置信区间(CI)为3.1 - 4.1)。在同性双胞胎中,RRR为14.0(95%CI为10.6 - 18.6),在异性双胞胎中,RRR为11.9(95%CI为7.1 - 19.9)。对于半同胞,RRR为1.5(95%CI为0.8 - 2.8)。当仅限于严重类型的先天性心脏病时,全同胞的RRR为6.9(95%CI为4.9 - 9.8)。在50%患有复发性先天性心脏病的同胞对中,同胞患有相似类型的先天性心脏病。复发的高相对风险表明家族风险因素在先天性心脏病的病因中很重要。

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