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

Katarina Bojanić,Marina Grubić,Toby N Weingarten,Ruža Grizelj,Lana Omerza,Juraj Sprung,Tomislav Ćaleta,Darrell R Schroeder,Jurica Vuković

doi:10.1186/s12955-018-0869-z

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.

Highlights

Patients with congenital diaphragmatic hernia (CDH) have a high residual morbidity rate
Using Pediatric Quality of Life Inventory (PedsQL), we found that the health-related quality of life (HRQoL) of the patients with CDH, as measured by the child self-reports and parent-proxy reports, was considerably lower than that of the control participants
Respiratory disease was the only characteristic markedly associated with low HRQoL in the child self-reports, but several characteristics—all descriptors of postnatal CDH severity—were associated with lower HRQoL when reported in the parent-proxy reports

Summary

Introduction

Patients 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. One study reported that 6- to 16-yearold patients with CDH have lower HRQoL than aged healthy children [14]. In a cross-sectional study that included patients born from 1969 through 1996, Poley et al [19] found that these differences in HRQoL between patients with CDH and healthy control patients decreased with age. Given the cross-sectional nature of that study, the enrolled patients were born when the primary treatment of CDH was considerably different than current treatment; differences among age groups could be due to survival bias or changes in treatment modalities. This rare patient population is most commonly examined using cross-sectional studies, and contemporary cohorts consisting of patients with CDH born after the introduction of new therapeutic modalities (eg, protective ventilation, extracorporeal membrane oxygenation [20,21,22]) are needed

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