Michalski Adrian M, Richardson Sandra D, Browne Marilyn L, Carmichael Suzan L, Canfield Mark A, VanZutphen Alissa R, Anderka Marlene T, Marshall Elizabeth G, Druschel Charlotte M
New York State Department of Health, Congenital Malformations Registry, Albany, New York.
Am J Med Genet A. 2015 May;167A(5):1071-81. doi: 10.1002/ajmg.a.36865. Epub 2015 Feb 25.
A small number of population-based studies have examined sex differences among infants with birth defects. This study presents estimates of sex ratio for both isolated cases and those with multiple congenital anomalies, as well as by race/ethnicity. Male-female sex ratios and their 95% confidence intervals were calculated for 25,952 clinically reviewed case infants included in the National Birth Defects Prevention Study (1997-2009), a large population-based case-control study of birth defects. The highest elevations in sex ratios (i.e., male preponderance) among isolated non-cardiac defects were for craniosynostosis (2.12), cleft lip with cleft palate (2.01), and cleft lip without cleft palate (1.78); the lowest sex ratios (female preponderance) were for choanal atresia (0.45), cloacal exstrophy (0.46), and holoprosencephaly (0.64). Among isolated cardiac defects, the highest sex ratios were for aortic stenosis (2.88), coarctation of the aorta (2.51), and d-transposition of the great arteries (2.34); the lowest were multiple ventricular septal defects (0.52), truncus arteriosus (0.63), and heterotaxia with congenital heart defect (0.64). Differences were observed by race/ethnicity for some but not for most types of birth defects. The sex differences we observed for specific defects, between those with isolated versus multiple defects, as well as by race/ethnicity, demonstrate patterns that may suggest etiology and improve classification.
少数基于人群的研究调查了出生缺陷婴儿的性别差异。本研究给出了孤立病例和患有多种先天性异常的病例的性别比估计值,以及按种族/族裔划分的性别比估计值。对纳入国家出生缺陷预防研究(1997 - 2009年)的25952例经过临床检查的病例婴儿计算了男女比例及其95%置信区间,该研究是一项基于人群的大型出生缺陷病例对照研究。孤立性非心脏缺陷中性别比最高(即男性占优势)的是颅缝早闭(2.12)、唇腭裂(2.01)和单纯唇裂(1.78);性别比最低(女性占优势)的是后鼻孔闭锁(0.45)、泄殖腔外翻(0.46)和前脑无裂畸形(0.64)。在孤立性心脏缺陷中,性别比最高的是主动脉狭窄(2.88)、主动脉缩窄(2.51)和大动脉d型转位(2.34);最低的是多发性室间隔缺损(0.52)、动脉干(0.63)和先天性心脏病合并内脏异位(0.64)。在某些但并非大多数类型的出生缺陷中,观察到了种族/族裔差异。我们观察到的特定缺陷在孤立性与多发性缺陷之间以及按种族/族裔划分的性别差异,显示出可能提示病因并改进分类的模式。