Prevalence and mortality in children with congenital diaphragmatic hernia: a multicountry study.
Prevalence and mortality in children with congenital diaphragmatic hernia: a multicountry study.
复制标题
DOI：
10.1016/j.annepidem.2020.11.007
复制
发表时间：
2021-04
影响因子：
5.6
通讯作者：
International Clearinghouse for Birth Defects Surveillance and Research
中科院分区：
文献类型：
--
作者：
Politis MD;Bermejo-Sánchez E;Canfield MA;Contiero P;Cragan JD;Dastgiri S;de Walle HEK;Feldkamp ML;Nance A;Groisman B;Gatt M;Benavides-Lara A;Hurtado-Villa P;Kallén K;Landau D;Lelong N;Lopez-Camelo J;Martinez L;Morgan M;Mutchinick OM;Pierini A;Rissmann A;Šípek A;Szabova E;Wertelecki W;Zarante I;Bakker MK;Kancherla V;Mastroiacovo P;Nembhard WN;International Clearinghouse for Birth Defects Surveillance and Research
先天性腹股沟疝（CDH）是一种严重的出生缺陷，其特征是腹部器官疝入胸腔的畸形，与高死亡率相关。本研究的目的是使用国际出生缺陷监测和研究中心（ICBDSR）下属多个国家基于医院和人群的出生缺陷监测项目收集的数据，检查（1）CDH的总体患病率和（2）CDH婴儿的死亡率和生存趋势。来自ICBDSR成员的19个国家的25个基于医院和人群的监测项目提供了1974年至2015年期间的出生缺陷死亡率数据。进一步检查了2001年至2012年的患病率估计和死亡率，这一时期大多数方案都有最完整的数据。包括涉及活产、死产或因胎儿畸形而选择性终止妊娠的CDH病例。根据Poisson回归计算每个国家和登记类型的患病率和95%置信区间（CI），根据Kaplan-Meier乘积限值法计算累积死亡率和95% CI。进行联合点回归分析以评估时间趋势。总体而言，所有国家合并的CDH患病率为2.6/10，000总出生（95% CI：2.5-2.7），2001年至2012年略有增加（平均年百分比变化[AAPC]=0.47%）。CDH的总体死亡率为37.7%，基于医院的登记研究中涉及活产的死亡人数多于基于人群的登记研究（45.1%比33.8%）。死亡率随着时间的推移而下降（AAPC=-2.43%）。患有多种先天性异常和综合征的婴儿的1周死亡率（45.2%和40.8%）高于患有孤立缺陷的婴儿（28.6%）。在两种登记类型中，CDH导致的大多数死亡发生在2- 6天的婴儿中（36.3%，基于医院; 12.1%，基于人群）。CDH的患病率随着时间的推移而增加;尽管死亡率略有下降，但仍然很高，特别是在出生后的第一周，并且因登记类型而异。需要进一步的研究来制定措施和干预措施，以减少CDH婴儿的死亡。
Congenital diaphragmatic hernia (CDH), a severe birth defect characterized by a diaphragmatic malformation allowing herniation by abdominal organs into the thorax, is associated with high mortality. The purpose of our study was to examine (1) the overall CDH prevalence and (2) mortality and survival trends of infants with CDH using data collected by hospital- and population-based birth defects surveillance programs from multiple countries affiliated with the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR). Twenty-five hospital- and population-based surveillance programs in 19 countries from members of the ICBDSR provided birth defects mortality data between 1974 and 2015. Prevalence estimates and mortality rates from 2001 to 2012, a period in which the majority of the programs had the most complete data, were further examined. Included were CDH cases involving live births, stillbirths, or elective termination of pregnancy for fetal anomalies. Prevalence and 95% confidence intervals (CI) from Poisson regression and cumulative mortality rates and 95% CI from the Kaplan-Meier Product-Limit method were calculated for each country and registry type. Joinpoint regression analyses were conducted to assess time trends. Overall, the prevalence of CDH from all countries combined was 2.6 per 10,000 total births (95% CI: 2.5–2.7), slightly increasing between 2001 and 2012 (average annual percent change [AAPC]=0.47%). The overall percent mortality of CDH was 37.7%, with hospital-based registries having more deaths involving live births than population-based registries (45.1% compared to 33.8%). Mortality rates decreased over time (AAPC=−2.43%). Infants with multiple congenital anomalies and syndromes had higher 1-week mortality rates (45.2% and 40.8%) than those with isolated defects (28.6%) overall. Most deaths due to CDH occurred among 2- to 6-day-old infants for both registry types (36.3%, hospital-based; 12.1%, population-based). The prevalence of CDH has increased over time; although the mortality rate has slightly decreased, it remains high especially during the first week of life and varied by registry type. Further research is needed to inform development of measures and interventions to decrease deaths among infants with CDH.
登录
查看更多内容
影响因子：
1.8
作者：
Al-Shanafey, S;Giacomantonio, M;Henteleff, H
通讯作者：
Henteleff, H
影响因子：
2.4
作者：
HARRISON, MR;BJORDAL, RI;KNUTRUD, O
通讯作者：
KNUTRUD, O
影响因子：
2.4
作者：
Paoletti, Monica;Raffler, Gabriele;Zani, Augusto
通讯作者：
Zani, Augusto
影响因子：
2.4
作者：
LANGER, JC;FILLER, RM;SUPERINA, RA
通讯作者：
SUPERINA, RA
影响因子：
2.4
作者：
Brownlee, E. M.;Howatson, A. G.;Sabharwal, A. J.
通讯作者：
Sabharwal, A. J.