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先天性膈疝患儿的自我报告呼吸和胃肠道结局:一项前瞻性多中心研究。

Self-reported respiratory and gastrointestinal outcomes in children with isolated congenital diaphragmatic hernia: A prospective multicentre study.

机构信息

Department of Development and Regeneration, Biomedical Sciences, KU Leuven, Leuven, Belgium.

BCNatal, Centre for Maternal-Fetal Medicine and Neonatology, Hospital Clínic and Hospital Sant Joan de Deu, University of Barcelona, IDIBAPS, IRSJD and Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Barcelona, Spain.

出版信息

BJOG. 2024 Oct;131(11):1506-1514. doi: 10.1111/1471-0528.17836. Epub 2024 May 15.

DOI:10.1111/1471-0528.17836
PMID:38747110
Abstract

OBJECTIVE

To evaluate medium-term self-reported respiratory and gastrointestinal (GI) outcomes in children with congenital diaphragmatic hernia (CDH).

DESIGN

Self-reported respiratory and GI outcomes correlated with prenatal severity indicators.

SETTING

Prospective study at three fetal medicine units.

POPULATION

Families of children prenatally diagnosed with isolated, left-sided CDH surviving for >1 year.

METHODS

Families received validated questionnaires for GI outcomes (Infant Gastroesophageal Reflux Questionnaire Revised, I-GERQ-R, for infants aged <2 years, or Paediatric Gastro-oesophageal Symptom and Quality of Life Questionnaire, PGSQ, for children aged aged 2-8 years or >9 years) and respiratory outcomes (preschool respiratory outcome questionnaire, for children aged ≤5 years, or the International Study of Asthma and Allergies in Childhood asthma questionnaire, for children aged 6-8 years or ≥9 years). Prenatal data collected from the medical records included lung size (percentage observed/expected lung-to-head ratio, O/E LHR %), liver position, fetal endoluminal tracheal occlusion (FETO) gestational age (GA) at delivery, and perinatal data included birthweight, location, patch repair and respiratory support.

MAIN OUTCOME MEASURES

The GI and respiratory scores were correlated with O/E LHR using linear and logistic regression models. Univariate analysis was used to evaluate associations with perinatal variables.

RESULTS

We obtained 142 responses from 342 families (representing a response rate of 45%). The baseline characteristics of participants and non-participants were comparable. No correlations between perinatal variables and respiratory or GI scores were identified. Children aged ≤5 years with lower O/E LHR values reported higher respiratory scores (P = 0.0175); this finding was not reported in older children. Overall, the children who underwent FETO (n = 51) had GI (P = 0.290) and respiratory (P = 0.052) scores that were comparable with those of children who were expectantly managed.

CONCLUSIONS

Families and children with prenatally diagnosed CDH reported fewer respiratory symptoms with increasing age. There was no correlation between O/E LHR or the use of FETO and self-reported outcomes.

摘要

目的

评估先天性膈疝(CDH)患儿的中期自我报告呼吸和胃肠道(GI)结局。

设计

自我报告的呼吸和 GI 结果与产前严重程度指标相关。

地点

三个胎儿医学单位的前瞻性研究。

人群

患有孤立性左侧 CDH 且存活时间>1 年的患儿的家庭。

方法

家庭接受了经过验证的 GI 结果问卷(<2 岁的婴儿使用婴儿胃食管反流问卷修订版(I-GERQ-R),2-8 岁或>9 岁的儿童使用儿科胃食管症状和生活质量问卷(PGSQ))和呼吸结果问卷(≤5 岁的儿童使用学龄前呼吸结局问卷,6-8 岁或≥9 岁的儿童使用国际儿童哮喘和过敏研究哮喘问卷)。从病历中收集的产前数据包括肺大小(观察到的/预期肺头比,O/E%LHR%)、肝脏位置、胎儿腔内气管阻塞(FETO)分娩时的胎龄(GA)和围产期数据,包括出生体重、位置、补片修复和呼吸支持。

主要观察指标

使用线性和逻辑回归模型将 GI 和呼吸评分与 O/E%LHR 相关联。使用单变量分析评估与围产期变量的关联。

结果

我们从 342 个家庭中获得了 142 个回复(代表 45%的回复率)。参与者和非参与者的基线特征相似。未发现围产期变量与呼吸或 GI 评分之间存在相关性。O/E%LHR 值较低的≤5 岁儿童报告的呼吸评分较高(P=0.0175);这一发现在年龄较大的儿童中并未报告。总体而言,接受 FETO 治疗的患儿(n=51)的 GI(P=0.290)和呼吸(P=0.052)评分与期待治疗的患儿相似。

结论

产前诊断为 CDH 的患儿及其家庭随着年龄的增长报告的呼吸症状较少。O/E%LHR 或 FETO 的使用与自我报告的结果之间没有相关性。

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