Long-term survival of children born with congenital anomalies: A systematic review and meta-analysis of population-based studies.
Long-term survival of children born with congenital anomalies: A systematic review and meta-analysis of population-based studies.
复制标题
DOI：
10.1371/journal.pmed.1003356
复制
发表时间：
2020-09
期刊：
影响因子：
15.8
通讯作者：
Rankin J
中科院分区：
文献类型：
--
作者：
Glinianaia SV;Morris JK;Best KE;Santoro M;Coi A;Armaroli A;Rankin J
随着传染病导致的全球儿童死亡率下降，先天性异常对儿童死亡率的相对贡献正在增加。尽管近几十年来，许多先天异常儿童的婴儿存活率有所改善，但有关婴儿期后存活率的证据较少。我们的目的是系统地回顾、总结和量化现有的基于人群的关于患有特定主要先天性异常的个体的长期生存的数据，并检查与生存相关的因素。检索了七个电子数据库（Medline、Embase、Scopus、PsycINFO、CINAHL、ProQuest Natural 和 Biological Science Collections）、参考文献列表以及 1995 年 1 月 1 日至 2020 年 4 月 30 日发表的研究中所包含文章的引文。资格筛选、数据提取和质量评估均重复进行。我们纳入了基于人群的原始研究，这些研究报告了患有严重先天性异常的儿童的长期生存（超过 1 年），并从出生开始进行随访，这些研究以英文形式发表为同行评审论文。由于最近对基于人群的 CHD 生存研究进行了系统回顾，因此排除了先天性心脏病 (CHD) 的研究。进行荟萃分析以汇集生存估计，解释随时间变化的趋势。在 10,888 篇已确定的文章中，55 篇（n = 367,801 例活产）符合纳入标准并进行了叙述性总结，41 项研究 (n = 54,676) 调查了八种先天性异常类型（脊柱裂 [n = 7,422]、脑膨出 [n = 1,562]、食管闭锁 [n = 6,303]、胆道闭锁 [n = 3,877]、膈疝 [n = 6,176]、腹裂 [n = 4,845]、伴有 CHD 的唐氏综合症 [n = 22,317] 和 18 三体症 [n = 2,174]）均纳入荟萃分析。这些研究涵盖了 1970 年至 2015 年的出生年份。随着时间的推移，患有脊柱裂、食道闭锁、胆道闭锁、膈疝、腹裂和唐氏综合症并伴有先心病的儿童的生存率显着提高，出生年份每 10 年增加的合并比值比 (OR) 为 OR = 1.34（95% 置信区间 [95%] CI] 1.24–1.46)，OR = 1.50 (95% CI 1.38–1.62)，OR = 1.62 (95% CI 1.28–2.05)，OR = 1.57 (95% CI 1.37–1.81)，OR = 1.24 (95% CI 1.02–1.5)，OR = 1.99 （95% CI 1.67–2.37）（除腹裂外，所有 p < 0.001 [p = 0.029]）。脑膨出儿童（OR = 0.98，95% CI 0.95–1.01，p = 0.19）和胆道闭锁儿童（OR = 0.96，95% CI 0.88–1.03，p = 0.26）没有观察到改善。额外的结构异常、低出生体重和早产是任何先天性异常类型生存率降低的最常见预测因素。荟萃分析的主要限制是研究数量少且队列规模小，这限制了模型的预测能力，导致置信区间较宽。这项系统回顾和荟萃分析总结了与主要先天性异常相关的长期生存的估计。我们报告了过去几十年来患有特定先天异常的儿童的生存率显着提高，并预测 2020 年出生的儿童的生存期估计可达 20 岁。这些信息对于规划和提供专门的医疗、社会和教育服务以及为受影响的家庭提供咨询非常重要。该试验已在 PROSPERO 数据库（CRD42017074675）上注册。在一项系统回顾和荟萃分析中，Svetlana V. Glinianaia 及其同事报告了患有特定先天异常的儿童长期生存的时间趋势。随着传染病导致的全球儿童死亡率下降，先天性异常对全球 5 岁以下儿童死亡率的相对贡献正在增加。识别和解决新出现的先天异常（包括 5-9 岁儿童）的优先事项是 2015 年后儿童健康议程的战略方向之一。本研究旨在总结和量化现有的基于人群的证据，证明患有童年时期表现出的特定主要先天性异常的儿童的长期生存率。这项系统回顾包括 55 项国际研究，这些研究估计了出生时患有严重先天异常的儿童 1 岁以上的生存率。我们对 1970 年至 2015 年出生年份的 41 项研究的荟萃分析结果显示，随着时间的推移，患有脊柱裂、食道闭锁、胆道闭锁、先天性膈疝、腹裂和与先天性心脏缺陷相关的唐氏综合症的儿童的生存率有统计学意义的显着改善，但没有证据表明患有脑膨出或具有天然肝脏的胆道闭锁的儿童的生存率有所改善。对于任何先天性异常类型来说，生存率降低的最常见的显着独立预测因素是存在额外的结构异常、低出生体重和较早的出生年份。过去几十年来，个别研究报告并经荟萃分析确定，患有特定先天异常的儿童的生存率显着提高，这对公共卫生、医疗、社会和家庭具有重要影响。有关 20 岁以下先天性异常儿童的预测存活率的信息对于为这些儿童规划专门的医疗、社会和教育服务以及估算与儿童和成年期特殊护理需求相关的费用非常重要。
Following a reduction in global child mortality due to communicable diseases, the relative contribution of congenital anomalies to child mortality is increasing. Although infant survival of children born with congenital anomalies has improved for many anomaly types in recent decades, there is less evidence on survival beyond infancy. We aimed to systematically review, summarise, and quantify the existing population-based data on long-term survival of individuals born with specific major congenital anomalies and examine the factors associated with survival. Seven electronic databases (Medline, Embase, Scopus, PsycINFO, CINAHL, ProQuest Natural, and Biological Science Collections), reference lists, and citations of the included articles for studies published 1 January 1995 to 30 April 2020 were searched. Screening for eligibility, data extraction, and quality appraisal were performed in duplicate. We included original population-based studies that reported long-term survival (beyond 1 year of life) of children born with a major congenital anomaly with the follow-up starting from birth that were published in the English language as peer-reviewed papers. Studies on congenital heart defects (CHDs) were excluded because of a recent systematic review of population-based studies of CHD survival. Meta-analysis was performed to pool survival estimates, accounting for trends over time. Of 10,888 identified articles, 55 (n = 367,801 live births) met the inclusion criteria and were summarised narratively, 41 studies (n = 54,676) investigating eight congenital anomaly types (spina bifida [n = 7,422], encephalocele [n = 1,562], oesophageal atresia [n = 6,303], biliary atresia [n = 3,877], diaphragmatic hernia [n = 6,176], gastroschisis [n = 4,845], Down syndrome by presence of CHD [n = 22,317], and trisomy 18 [n = 2,174]) were included in the meta-analysis. These studies covered birth years from 1970 to 2015. Survival for children with spina bifida, oesophageal atresia, biliary atresia, diaphragmatic hernia, gastroschisis, and Down syndrome with an associated CHD has significantly improved over time, with the pooled odds ratios (ORs) of surviving per 10-year increase in birth year being OR = 1.34 (95% confidence interval [95% CI] 1.24–1.46), OR = 1.50 (95% CI 1.38–1.62), OR = 1.62 (95% CI 1.28–2.05), OR = 1.57 (95% CI 1.37–1.81), OR = 1.24 (95% CI 1.02–1.5), and OR = 1.99 (95% CI 1.67–2.37), respectively (p < 0.001 for all, except for gastroschisis [p = 0.029]). There was no observed improvement for children with encephalocele (OR = 0.98, 95% CI 0.95–1.01, p = 0.19) and children with biliary atresia surviving with native liver (OR = 0.96, 95% CI 0.88–1.03, p = 0.26). The presence of additional structural anomalies, low birth weight, and earlier year of birth were the most commonly reported predictors of reduced survival for any congenital anomaly type. The main limitation of the meta-analysis was the small number of studies and the small size of the cohorts, which limited the predictive capabilities of the models resulting in wide confidence intervals. This systematic review and meta-analysis summarises estimates of long-term survival associated with major congenital anomalies. We report a significant improvement in survival of children with specific congenital anomalies over the last few decades and predict survival estimates up to 20 years of age for those born in 2020. This information is important for the planning and delivery of specialised medical, social, and education services and for counselling affected families. This trial was registered on the PROSPERO database (CRD42017074675). In a systematic review and meta-analysis, Svetlana V. Glinianaia and colleagues report on temporal trends in long-term survival of children born with specific congenital anomalies. Following a reduction in global child mortality due to communicable diseases, the relative contribution of congenital anomalies to child mortality under age 5 years is increasing globally. Identifying and addressing the emerging priority of congenital anomalies, including for children aged 5–9 years, is one of the strategic directions for the post-2015 child health agenda. This research aimed to summarise and quantify the existing population-based evidence on long-term survival of children born with specific major congenital anomalies that manifest in childhood. This systematic review included 55 international studies that estimated survival beyond 1 year of age of children born with major congenital anomalies. Our meta-analysis results of 41 studies over the birth years 1970–2015 showed a statistically significant improvement in survival over time in children with spina bifida, oesophageal atresia, biliary atresia, congenital diaphragmatic hernia, gastroschisis, and Down syndrome associated with a congenital heart defect, but there was no evidence of improvement in those with encephalocele or biliary atresia with a native liver. The commonest significant independent predictors of reduced survival for any congenital anomaly type were presence of additional structural anomalies, low birth weight, and earlier birth year period. A significant improvement in survival of children with specific congenital anomalies over the last few decades reported by individual studies and identified by the meta-analysis has important public health, medical, social, and family implications. Information on predicted survival of children with congenital anomalies up to 20 years of age is important for planning specialised medical, social, and education services for these children and for estimating costs associated with special care needs in childhood and adulthood.
登录
查看更多内容
影响因子：
3.6
作者：
Eide, MG;Skærven, R;Oyen, N
通讯作者：
Oyen, N
影响因子：
12.6
作者：
de Vries, Willemien;Homan-Van der Veen, Jenneke;Verkade, Henkjan J.
通讯作者：
Verkade, Henkjan J.
影响因子：
9.8
作者：
Bagos, Pantelis G.
通讯作者：
Bagos, Pantelis G.
DOI：
10.1002/bdra.20218
发表时间：
2006-01-01
影响因子：
--
作者：
Agha, MM;Williams, JI;Dodds, L
通讯作者：
Dodds, L
影响因子：
5.2
作者：
Dastgiri, S;Gilmour, WH;Stone, DH
通讯作者：
Stone, DH