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1994 年至 2021 年期间丹麦活产儿中先天性腹裂的流行情况和伴发畸形:一项全国范围内基于登记的研究。

Gastroschisis Prevalence and Co-occurring Malformations Among Danish Live Births During 1994-2021: A Nationwide Register-Based Study.

机构信息

Department of Neonatology, University Hospital Rigshospitalet, Copenhagen, Denmark; Department for Congenital Disorders, Statens Serum Institute, Copenhagen, Denmark.

Department for Congenital Disorders, Statens Serum Institute, Copenhagen, Denmark; Department of Epidemiology, College of Public Health, The University of Iowa, Iowa City, IA, USA.

出版信息

J Pediatr Surg. 2024 Dec;59(12):161931. doi: 10.1016/j.jpedsurg.2024.161931. Epub 2024 Sep 14.

Abstract

BACKGROUND

Gastroschisis prevalence has increased for decades with corresponding increases in the need for immediate and follow-up care. Such care can be complicated by presence of co-occurring malformations. This study explores prevalence of gastroschisis and co-occurring malformations among a 28-year cohort of Danish live-born infants.

METHODS

This retrospective cohort study used data from 1,695,992 infants born in Denmark during 1994-2021 and registered in the neonatal screening program. Infants were identified from the Danish Civil Registration System and Danish National Patient Register accessed through the Danish Biobank Register. Data on co-occurring malformations were ascertained to classify infants as syndromic or non-syndromic (either isolated or with co-occurring major malformations) and on selected infant and parental characteristics. Poisson regression models were used to estimate prevalence and corresponding 95% confidence intervals (CIs).

RESULTS

Prevalence (per 10,000 live births) of gastroschisis was 1.64 (CI: 1.45-1.84). Temporal trend analyses showed a statistically significant annual increase of 2.8% (CI: 1.4-3.3). Infants with gastroschisis most often presented as isolated (77.7%; CI: 72.3-82.5), followed by those with co-occurring malformations (21.9%; CI: 17.2-27.3) or a diagnosed syndrome (0.4%, CI: <0.1-2.0). Among infants with co-occurring malformations, cardiovascular (10.9%; CI: 6.8; 12.2) and intestinal (9.0%; CI: 5.9-12.2) malformations were most frequently recorded. Prevalence was higher among infants classified as premature but not influenced by infant sex or parental nativity.

CONCLUSION

Gastroschisis prevalence in Denmark increased during 1994-2021, similar to international reports, without increase in co-occurring malformations. Future work with this cohort will characterize healthcare received, comorbidities, and outcomes across the lifespan.

LEVELS OF EVIDENCE

Level III (High-quality prospective cohort study).

摘要

背景

几十年来,先天性腹裂的发病率一直在上升,随之而来的是对即时和后续护理的需求增加。这种护理可能会因同时存在的畸形而变得复杂。本研究探讨了丹麦 28 年活产婴儿队列中先天性腹裂的发病率和同时存在的畸形。

方法

本回顾性队列研究使用了 1994 年至 2021 年期间在丹麦出生的 1695992 名婴儿的数据,这些数据登记在新生儿筛查计划中。通过丹麦民事登记系统和丹麦国家患者登记处从丹麦生物银行登记处获得婴儿数据。通过分类为综合征或非综合征(孤立或同时存在主要畸形)以及特定婴儿和父母特征来确定同时存在的畸形数据。使用泊松回归模型估计发病率和相应的 95%置信区间(CI)。

结果

先天性腹裂的发病率(每 10000 例活产)为 1.64(CI:1.45-1.84)。时间趋势分析显示,发病率呈统计学意义的每年 2.8%的增长(CI:1.4-3.3)。患有先天性腹裂的婴儿最常见的是孤立的(77.7%;CI:72.3-82.5),其次是同时存在畸形的(21.9%;CI:17.2-27.3)或诊断为综合征的(0.4%;CI:<0.1-2.0)。同时存在畸形的婴儿中,心血管畸形(10.9%;CI:6.8;12.2)和肠道畸形(9.0%;CI:5.9-12.2)最常被记录。在早产儿中发病率较高,但不受婴儿性别或父母出生地的影响。

结论

1994 年至 2021 年期间,丹麦先天性腹裂的发病率增加,与国际报告相似,同时存在的畸形没有增加。未来将对该队列进行研究,以描述终生的医疗保健、合并症和结局。

证据水平

三级(高质量前瞻性队列研究)。

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