Health-related quality of life in children and adolescents with congenital diaphragmatic hernia: a cross-sectional study

Abstract

BackgroundPatients with congenital diaphragmatic hernia (CDH) have a high residual morbidity rate. We compared self-reported health-related quality of life (HRQoL) between patients with CDH and healthy children.MethodsForty-five patients with CDH who were born from January 1, 1990, through February 15, 2015, were matched to healthy, age-matched control participants at a 1:2 ratio. The health records of the study participants were reviewed to determine comorbid conditions, and HRQoL was assessed by both the participants and their parents with the Pediatric Quality of Life Inventory (PedsQL). The HRQoL scores of the patients with CDH and the control participants were compared by using analysis of variance to adjust for age group and sex. Among patients with CDH, analysis of variance was used to compare HRQoL scores across groups defined according to their characteristics at initial hospitalization, postdischarge events, and comorbid conditions.ResultsCompared with control participants, patients with CDH had lower mean PedsQL scores, as reported by the parent and child, for the physical and psychosocial domains (P < 0.001). Risk factors associated with lower parent-reported HRQoL included bronchopulmonary dysplasia, longer initial hospitalization, severe cognitive impairment, and orthopedic symptoms; among patients with CDH, low HRQoL was associated with chronic respiratory issues.ConclusionPatients with CDH had lower HRQoL compared with healthy participants. Parent-reported HRQoL tended to be higher than child-reported HRQoL. Results were also inconsistent for the risk factors associated with HRQoL obtained by using child- and parent-reported scores. Therefore, when interpreting HRQoL in CDH survivors, a proxy report should not be considered a substitute for a child’s self-report.

Long-Term Health-Related Quality of Life in Survivors of Congenital Diaphragmatic Hernia

“…The two studies that reported a lower HRQoL in CDH survivors, Bojanić et al and Tan et al, were the only studies to use age-matched controls rather than normative data. 14,39 The study by Bojanić et al compared 45 CDH children with a mean age of 8.2 years with 90 age-matched controls without chronic diseases. 39 They used the PedsQL questionnaires for patients and parents.…”

“…14,39 The study by Bojanić et al compared 45 CDH children with a mean age of 8.2 years with 90 age-matched controls without chronic diseases. 39 They used the PedsQL questionnaires for patients and parents. Patients and parents scored patients significantly lower than controls in physical and psychosocial domains.…”

“…Bojanić et al subclassified patients into "low or intermediate" and "high" probability of survival based on the Congenital Diaphragmatic Hernia Study Group survival score 41 ; there was no difference in the HRQoL. 39 Bojanić et al reported lower parent-reported HRQoL with bronchopulmonary dysplasia, longer hospitalization, severe cognitive impairment, and orthopaedic symptoms. Lower patient-reported HRQoL was only associated with chronic respiratory issues.…”

“…Bojanić et al's study suggesting a lower HRQoL for CDH survivors compared patients with healthy age-matched controls. 39 Children with chronic medical conditions were specifically excluded from the control cohort, and this may have inflated the control HRQoL. The four studies that compared patients to normative values included all children regardless of health status; therefore, the methodology that could have been a strength may have hampered Bojanić et al's results.…”

“…40 The CDH Study Group disease severity score determined at 5 minutes of life did not predict HRQoL. 39 Mosberger et al's study found that the median HRQoL score was significantly higher after thoracoscopic repair versus more invasive surgical methods, suggesting that thoracoscopic repair is advantageous to HRQoL. 42 This conclusion is biased as only less severe patients are treated with thoracoscopy; therefore, disease severity and not surgical methodology may be the influencing factor.…”