Longitudinal Health Status and Quality of Life After Esophageal Atresia Repair

Abstract

Objectives: To longitudinally evaluate self-reported and proxy-reported health status (HS) and quality of life (QoL) of school-aged children born with esophageal atresia (EA). Methods: We obtained Pediatric Quality of Life Inventory (HS) and DUX-25 (QoL) questionnaires from children born with EA between 1999 and 2011 at 8 and/or 12 years old. Children completed self-reports during neuropsychological assessments in a prospective longitudinal follow-up program. Parents filled out proxy-reports at home. Total and subscale scores were evaluated longitudinally and compared with sex-specific reference norms. Results: In total, 110 participants (62% boys) were included. Self-reported HS improved significantly between 8 and 12 years for both boys (mean difference [md] 4.35, effect size [ES] 0.54, P ¼ 0.009) and girls (md 3.26, ES 0.63, P ¼ 0.004). Proxy-reported HS tended to improve over time, while self-reported and proxy-reported QoL tended to decline. Self-reported HS at 8 years was below normal for both boys (md À5.44, ES À0.35, P < 0.001) and girls (md À7.61, ES À0.32, P < 0.001). Girls' self-reported QoL was below normal at 8 (md À5.00, ES À0.18, P ¼ 0.019) and 12 years (md À10.50, ES À0.26, P ¼ 0.001). Parents reported normal HS at both ages, whereas they rated the QoL of their daughters below normal at 12 years (md À10.00, ES À0.16, P ¼ 0.022). All above results are total scores. Conclusions: Self-reported and proxy-reported HS of children with EA improved between 8 and 12 years, while their QoL tended to decline. We recommend to consider HS and QoL as two separate concepts and to measure both simultaneously and longitudinally when evaluating the burden of disease.

Psychometric Performance of a Condition-Specific Quality-of-Life Instrument for Dutch Children Born with Esophageal Atresia

“…Furthermore, the parental educational level was higher than in the general population. Although this is a common finding in the EA population [ 12 , 48 ] and in psychometric evaluation in general [ 49 ], it should be taken into account that this could lead to bias. Moreover, the online study set-up and some statistical assumptions differed from earlier EA-QOL© validation studies.…”

“…Despite inconsistent findings, studies indicated that children with EA report lower generic HRQoL than do healthy children [ 9 , 10 , 11 , 12 ]. Condition-specific instruments tend to be more sensitive to the detection and discrimination of clinical morbidities, and more suitable for assessing disease burden [ 13 ].…”

The DUX-25 after Twenty-Five Years: New Analyses and Reference Data

“…This is relevant in that the DUX-25 focuses on a client’s emotional and cognitive evaluation to measure HRQOL, while the PedsQL uses functioning as an indicator. The main difference between these concepts is that while functioning attempts to measure well-being by defining specific concepts externally and objectively, the subjective emotional and cognitive evaluation indicates how an individual actually experiences limitations in specific areas of his or her life [ 41 ]. In certain clinical groups, the subjective and the more objective view of health can produce different results over time, as we have already substantiated in a study in children after oesophageal atresia repair [ 20 ].…”

Swallowing-related quality of life in children with oesophageal atresia: a national cohort study

“…Herewith, we confirm previous study results, where young children with OA (< 5 years) are affected by any kind of feeding or swallowing problems in about 55–70% of cases, compared to only about 18–30% of older children with OA [ 2 , 8 , 16 , 31 ]. In a recent study from the Netherlands, self-reported health status in children with OA was also increasing with age [ 32 ]. These changes with age might be due to an accommodation of the swallowing ability and integration of feeding disorders by time [ 15 ].…”

“…VACTERL is a combination of various birth defects, such as vertebral defects, anorectal malformations, cardiac defects, OA, tracheo-oesophageal fistula and renal and limb malformations which can cause physical dysfunction. Self-perceived health status in children with OA and VACTERL is also reported to be worse compared to children with OA only [ 32 ]. VACTERL associated life-long conditions such as neurodevelopmental delay and attention deficit may lead to abnormal eating habits.…”