Disease severity impacts perceived quality of life in congenital diaphragmatic hernia: a prospective observational study

Author:

Dimmer AlexandraORCID,Meehan Madison,Beauseigle Sabrina,Koclas Louise,Paquette Katryn,Michel Macias CarolinaORCID,Moore Shiran S,Sant'Anna Ana,Shapiro Adam,Simoneau Jessica,Villegas Martinez Daniela,Altit Gabriel,Puligandla PramodORCID

Abstract

BackgroundWhile research indicates comparable quality of life (QOL) in congenital diaphragmatic hernia (CDH) and healthy populations, the effect of CDH severity on patients’ health perceptions remains unexplored. We aimed to assess QOL perception in CDH, hypothesising a decline correlated with increased disease severity.MethodsIn this prospective observational study, we analysed patients with CDH aged 5 years and above participating in a longitudinal outpatient programme. We excluded bilateral CDH, genetic/syndromic conditions, prematurity and late diagnosis. Participants self-administered the age-adapted Pediatric Quality of Life Inventory (PedsQL) survey, covering four domains (physical, emotional, social, school). After enrolment, data were collected blind to severity status (larger defects denoting significant/‘severe’ disease). Repeated measurements were managed using a random mixed-effects model.ResultsOf 34 participants (50% males) who completed the PedsQL, 10 provided measurements at two visits. Eight required a patch (type C), while 26 had primary repairs (type A=8; type B=18). Age at first evaluation was comparable across groups (no patch: median 11 (7–16), patch: 13 (8–15) years, p=0.78). Severe CDH correlated significantly with lower PedsQL scores (adjusted β: −18%, 95% CI −28%; −7%, adjusted for age at visit and sex). Lower scores specifically occurred in walking, exercising, social and academic functioning.ConclusionSevere CDH significantly lowers QOL. This finding is crucial for resource allocation in long-term CDH health surveillance and advocates for regular inclusion of patient experiences in quality improvement efforts.

Publisher

BMJ

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