Neurodevelopment and Health-Related Quality of Life in Infants Born with Gastroschisis: A 6-Year Retrospective French Study

Abstract

Introduction Quantify quality of life (QoL) outcomes in gastroschisis children is little assessed. The primary objective was to describe the long-term outcome of newborns with gastroschisis treated in three tertiary care hospitals of France in terms of neurodevelopment and QoL. Materials and Methods The study reported was a cross-sectional, descriptive multicentric retrospective study assessing the outcome of newborns with gastroschisis, born between January 1, 2009, and December 31, 2014, treated at two large and French level III neonatal intensive care units. Long-term outcome data were assessed by questionnaires sent to the infants' parents. Questionnaires explored global health, neurological development, and quality of life (overall assessment including socio-economic and medical), Age & Stages Questionnaire, infants' quality of life (KIDSCREEN), and quality of parents' life (General Questionnaire Short Form-36). Results In this study, 50% of the survivor's families answered the assessment form (n = 33). The average follow-up age was 40 months, ranging from 8 months to 6 years. Cases of simple gastroschisis more often had a normal score for “communication” (p = 0.033), while patients who received morphine for a longer duration had significantly lower scores for the items “communication” and “problem resolving” (p = 0.024 and p = 0.011, respectively). Children's QoL was significantly lower for patients with gestational age younger than 36 weeks (p = 0.023) and for patients born following “fetal cause delivery” (p = 0.022). Parents had a significantly higher physical composite score if their child underwent primary closure (p = 0.012). Conclusion Our analyses confirm the idea that cases of complex gastroschisis and preterm delivery may lead to poorer outcome. Such hindsight (40 months in mean) allowed for an interesting assessment of development long after the patient's initial hospitalization and to confirm these results, a standardized neuropsychological evaluation of patients should be done when at least 6 years old. An accurate assessment of the social environment and its impact on the development and QoL of children will be fundamental to avoid selection bias.

Long-Term Neurodevelopmental Outcomes in Children with Gastroschisis: A Review of the Literature

“…Ten studies were included in the school age group (6 years-17 years 11 months). 41,[58][59][60][61][62][63][64][65][66] Cognitive and intellectual development once again demonstrated conflicting findings, similar to results in the infant/toddler age range. Some studies (n ¼ 4) evaluating cohorts inclusive of children with GS found that overall intellectual functioning was consistent with population norms.…”

“…Quality of life Tosello et al 2017 65 vision outcomes and found they performed comparably to age norms. Data available on motor performance in heterogenous study cohorts inclusive of children with GS exhibited more contradictory results.…”

“…Motor functioning Gold et al 2020 59 Children's Global Assessment Emotional and behavioral functioning Tosello et al 2017 65 Parentreported surveys Piers-Harris Children's Self-Concept Scale…”

“…Emotional functioning Harris et al 2016 64 Infant's Quality of Life (KIDSCREEN) Quality of life Tosello et al 2017 65 Parents' Quality of Life-General Questionnaire Short Form (SF-36)…”

Gastroschisis for the Gastroenterologist: Updates on Epidemiology, Management, and Outcomes

“…Despite the delicate neonatal course and risk for repeat surgery, patients with gastroschisis tend to have neurodevelopmental outcomes which are similar to age- and weight-matched neonatal intensive care unit graduate controls at 1–2 years follow-up (28,29). Young children with history of complex gastroschisis demonstrate worse neurodevelopmental outcomes compared to those with simple gastroschisis (30). Despite worse short-term outcomes and more reported abdominal symptoms (ie, abdominal pain, bloating, diarrhea) in children with history of complex gastroschisis, parent-reported quality of life scores remain similar to children with history of simple gastroschisis (23).…”