Gastroschisis at school age: what do parents report?

Annelieke Hijkoop,André B Rietman,Dick Tibboel,Hanneke Ijsselstijn,René M H Wijnen,Titia E Cohen-Overbeek,Joost Van Rosmalen

doi:10.1007/s00431-019-03417-5

Abstract

Children with gastroschisis are at high risk of morbidity in early life, which could affect long-term outcomes. We determined parent-reported outcomes in school-aged children born in 2000–2012, using paper questionnaires. Parent-perceived child vulnerability and motor function were compared with the Dutch reference data; parent-rated data on cognition, health status, quality of life, and behavior were compared with those of controls matched for age, gender, and maternal education level. Of 77 eligible participants, 31 (40%) returned the questionnaires. Parent-reported motor function was normal in 23 (74%) children. Total scores on health status, quality of life, and behavior did not differ significantly from those of matched controls. Children with gastroschisis had lower scores on cognition (median (interquartile range); 109 (87–127)) than their matched controls (124 (113–140); p = 0.04). Neonatal intestinal failure and increased parent-perceived vulnerability were associated with lower scores on cognition (β − 25.66 (95% confidence interval − 49.41, − 1.91); − 2.76 (− 5.27, − 0.25), respectively).Conclusion: Parent-reported outcomes of school-aged children with gastroschisis were mainly reassuring. Clinicians and parents should be aware of the higher risk of cognitive problems, especially in those with neonatal intestinal failure or increased parent-perceived vulnerability. We recommend multidisciplinary follow-up at school age of children with gastroschisis and neonatal intestinal failure.What is Known:• Many infants with gastroschisis experience morbidity in early life.• Data on developmental outcomes and daily functioning in children with gastroschisis beyond the age of 5 years are scarce and conflicting.What is New:• Parents of school-aged children treated for gastroschisis report normal motor function, health status, quality of life, and behavior.• Children with gastroschisis, especially those with intestinal failure, may be at risk for cognitive problems at school age. Parents who reported their child as being more vulnerable also reported more cognitive problems at school age.

Highlights

Gastroschisis is a life-threatening congenital abdominal wall defect requiring surgical treatment shortly after birth
Clinicians and parents should be aware of the higher risk of cognitive problems, especially in those with neonatal intestinal failure or increased parentperceived vulnerability
Parents of school-aged children treated for gastroschisis report normal motor function, health status, quality of life, and behavior

Summary

Introduction

Gastroschisis is a life-threatening congenital abdominal wall defect requiring surgical treatment shortly after birth. Additional anomalies are relatively rare, and survival rates are over 90% [2]. These infants are at high risk of morbidity, especially those with associated intestinal defects (complex gastroschisis [3]). In addition to having undergone surgery in early life, many of these infants are born small for gestational age (SGA) [8, 9] or preterm [2], which may affect neurodevelopmental outcomes [10,11,12,13]. Twenty-eight (90%) were raised by at least one biological parent, and three (10%) lived in a foster family. Seven (23%) of 30 children required medication; one parent did not answer this question.

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Discussion

Conclusion