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Self-reported respiratory and gastrointestinal outcomes in children with isolated congenital diaphragmatic hernia: A
Mary Patrice Eastwood1,2, Lennart Van der Veeken1, Luc Joyeux1
1Department of Development and Regeneration, Biomedical Sciences, KU Leuven, Leuven, Belgium.
Insights
Medium-term respiratory and gastrointestinal outcomes in children with congenital diaphragmatic hernia (CDH) were evaluated. Respiratory symptoms decreased with age, with no correlation found between lung size or FETO use and self-reported outcomes.
Area of Science:
- Pediatric Surgery
- Neonatology
- Pulmonology
Background:
- Congenital diaphragmatic hernia (CDH) is a complex birth defect requiring specialized care.
- Understanding medium-term outcomes is crucial for improving patient management and long-term prognosis.
- Self-reported outcomes provide valuable insights into the patient's quality of life post-treatment.
Purpose of the Study:
- To assess medium-term self-reported respiratory and gastrointestinal (GI) symptoms in children diagnosed with congenital diaphragmatic hernia (CDH).
- To correlate these outcomes with prenatal indicators of disease severity, such as lung size and liver position.
- To evaluate the impact of interventions like fetal endoluminal tracheal occlusion (FETO) on patient-reported symptoms.
Main Methods:
- A prospective study was conducted across three fetal medicine units.
- Families of children with isolated, left-sided CDH surviving over one year completed validated questionnaires for respiratory and GI symptoms.
- Data on prenatal severity (O/E LHR, liver position) and perinatal factors were collected and analyzed using regression models.
Main Results:
- A 45% response rate was achieved from 342 families, with comparable characteristics between participants and non-participants.
- No significant correlations were found between perinatal variables and respiratory or GI scores.
- Children aged 5 years and younger with lower observed/expected lung-to-head ratio (O/E LHR) reported increased respiratory symptoms (P=0.0175).
- Children who underwent FETO (n=51) showed comparable GI and respiratory scores to those managed expectantly.
Conclusions:
- Self-reported respiratory symptoms in children with CDH tend to decrease with increasing age.
- Prenatal indicators like O/E LHR and interventions such as FETO did not show a significant correlation with medium-term self-reported respiratory or GI outcomes.
- These findings highlight the importance of age-related symptom progression and suggest that current prenatal severity markers may not fully predict long-term patient-reported well-being.
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.
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