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先天性无耳畸形和小耳畸形患儿伴发畸形的模式。

Patterns of co-occurring birth defects in children with anotia and microtia.

机构信息

Department of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.

Department of Epidemiology, Human Genetics & Environmental Sciences, UTHealth School of Public Health, Houston, Texas, USA.

出版信息

Am J Med Genet A. 2023 Mar;191(3):805-812. doi: 10.1002/ajmg.a.63081. Epub 2022 Dec 21.

Abstract

Many infants with anotia or microtia (A/M) have co-occurring birth defects, although few receive syndromic diagnoses in the perinatal period. Evaluation of co-occurring birth defects in children with A/M could identify patterns indicative of undiagnosed/unrecognized syndromes. We obtained information on co-occurring birth defects among infants with A/M for delivery years 1999-2014 from the Texas Birth Defects Registry. We calculated observed-to-expected ratios (OER) to identify birth defect combinations that occurred more often than expected by chance. We excluded children diagnosed with genetic or chromosomal syndromes from analyses. Birth defects and syndromes/associations diagnosed ≤1 year of age were considered. We identified 1310 infants with non-syndromic A/M, of whom 38% (N = 492) were diagnosed with co-occurring major defects. Top combinations included: hydrocephalus, ventricular septal defect, and spinal anomalies (OER 58.4); microphthalmia and anomalies of the aorta (OER 55.4); and cleft lip with or without cleft palate and rib or sternum anomalies (OER 32.8). Some combinations observed in our study may represent undiagnosed/atypical presentations of known A/M associations or syndromes, or novel syndromes yet to be described in the literature. Careful evaluation of infants with multiple birth defects including A/M is warranted to identify individuals with potential genetic or chromosomal syndromes.

摘要

许多患有无耳症或小耳症(A/M)的婴儿伴有其他出生缺陷,尽管在围产期很少被诊断为综合征。评估 A/M 患儿的其他出生缺陷可以发现未确诊/未识别综合征的模式。我们从德克萨斯州出生缺陷登记处获取了 1999 年至 2014 年期间患有 A/M 的婴儿的其他出生缺陷信息。我们计算了观察到的与预期的比值(OER),以确定出生缺陷组合发生的频率高于预期的随机概率。我们从分析中排除了诊断为遗传或染色体综合征的儿童。研究考虑了≤1 岁时诊断出的出生缺陷和综合征/关联。我们确定了 1310 名患有非综合征性 A/M 的婴儿,其中 38%(N=492)被诊断为伴有其他主要缺陷。主要组合包括:脑积水、室间隔缺损和脊柱异常(OER 58.4);小眼症和主动脉异常(OER 55.4);以及唇裂伴或不伴腭裂和肋骨或胸骨异常(OER 32.8)。我们研究中观察到的一些组合可能代表了已知 A/M 关联或综合征的未确诊/非典型表现,或者是尚未在文献中描述的新综合征。需要对伴有多种出生缺陷(包括 A/M)的婴儿进行仔细评估,以识别可能患有潜在遗传或染色体综合征的个体。

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