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.

Keywords

gastroschisis - neurodevelopmental - outcome - quality of life

• References

• 1 Prefumo F, Izzi C. Fetal abdominal wall defects. Best Pract Res Clin Obstet Gynaecol 2014; 28 (03) 391-402
• 2 Emil S, Canvasser N, Chen T, Friedrich E, Su W. Contemporary 2-year outcomes of complex gastroschisis. J Pediatr Surg 2012; 47 (08) 1521-1528
• 3 Grant NH, Dorling J, Thornton JG. Elective preterm birth for fetal gastroschisis. Cochrane Database Syst Rev 2013; 6 (06) CD009394
• 4 Olsen EA, Brambrink AM. Anesthetic neurotoxicity in the newborn and infant. Curr Opin Anaesthesiol 2013; 26 (05) 535-542
• 5 Harris EL, Hart SJ, Minutillo C. , et al. The long-term neurodevelopmental and psychological outcomes of gastroschisis: a cohort study. J Pediatr Surg 2016; 51 (04) 549-553
• 6 Carpenter JL, Wiebe TL, Cass DL, Olutoye OO, Lee TC. Assessing quality of life in pediatric gastroschisis patients using the Pediatric Quality of Life Inventory survey: an institutional study. J Pediatr Surg 2016; 51 (05) 726-729
• 7 Ravens-Sieberer U, Erhart M, Rajmil L. , et al; European KIDSCREEN Group. Reliability, construct and criterion validity of the KIDSCREEN-10 score: a short measure for children and adolescents' well-being and health-related quality of life. Qual Life Res 2010; 19 (10) 1487-1500
• 8 Leplège A, Ecosse E, Coste J, Pouchot J, Perneger T. Le questionnaire MOS SF-36: manuel de l'utilisateur et guide d'interprétation des scores. Paris: Editions Estem; 2001: 155
• 9 Logghe HL, Mason GC, Thornton JG, Stringer MD. A randomized controlled trial of elective preterm delivery of fetuses with gastroschisis. J Pediatr Surg 2005; 40 (11) 1726-1731
• 10 Carnaghan H, Baud D, Lapidus-Krol E. , et al. Effect of gestational age at birth on neonatal outcomes in gastroschisis. J Pediatr Surg 2016; 51 (05) 734-738
• 11 Tanis JC, Van Braeckel KN, Kerstjens JM, Bocca-Tjeertes IF, Reijneveld SA, Bos AF. Functional outcomes at age 7 years of moderate preterm and full term children born small for gestational age. J Pediatr 2015; 166 (03) 552-8.e1
• 12 Henrich K, Huemmer HP, Reingruber B, Weber PG. Gastroschisis and omphalocele: treatments and long-term outcomes. Pediatr Surg Int 2008; 24 (02) 167-173
• 13 van Manen M, Hendson L, Wiley M, Evans M, Taghaddos S, Dinu I. Early childhood outcomes of infants born with gastroschisis. J Pediatr Surg 2013; 48 (08) 1682-1687
• 14 Flamant C, Branger B, Nguyen The Tich S. , et al. Parent-completed developmental screening in premature children: a valid tool for follow-up programs. PLoS One 2011; 6 (05) e20004
• 15 Tunell WP, Puffinbarger NK, Tuggle DW, Taylor DV, Mantor PC. Abdominal wall defects in infants. Survival and implications for adult life. Ann Surg 1995; 221 (05) 525-528 , discussion 528–530
• 16 Skellern CY, Rogers Y, O'Callaghan MJ. A parent-completed developmental questionnaire: follow up of ex-premature infants. J Paediatr Child Health 2001; 37 (02) 125-129
• 17 Woodward BJ, Papile L-A, Lowe JR. , et al. Use of the Ages and Stages Questionnaire and Bayley Scales of Infant Development-II in neurodevelopmental follow-up of extremely low birth weight infants. J Perinatol 2011; 31 (10) 641-646
• 18 Rydz D, Srour M, Oskoui M. , et al. Screening for developmental delay in the setting of a community pediatric clinic: a prospective assessment of parent-report questionnaires. Pediatrics 2006; 118 (04) e1178-e1186
• 19 Harris EL, Minutillo C, Hart S. , et al. The long term physical consequences of gastroschisis. J Pediatr Surg 2014; 49 (10) 1466-1470
• 20 Dellschaft NS, Alexandre-Gouabau M-C, Gardner DS. , et al. Effect of pre- and postnatal growth and post-weaning activity on glucose metabolism in the offspring. J Endocrinol 2015; 224 (02) 171-182
• 21 Ross MG, Beall MH. Adult sequelae of intrauterine growth restriction. Semin Perinatol 2008; 32 (03) 213-218
• 22 Lepigeon K, Van Mieghem T, Vasseur Maurer S, Giannoni E, Baud D. Gastroschisis--what should be told to parents?. Prenat Diagn 2014; 34 (04) 316-326
• 23 Koivusalo A, Lindahl H, Rintala RJ. Morbidity and quality of life in adult patients with a congenital abdominal wall defect: a questionnaire survey. J Pediatr Surg 2002; 37 (11) 1594-1601
• 24 Michel F, Baumstarck K, Gosselin A. , et al; PACA Group Research for Quality of Life of children with a congenital diaphragmatic hernia. Health-related quality of life and its determinants in children with a congenital diaphragmatic hernia. Orphanet J Rare Dis 2013; 8: 89
• 25 Batorowicz B, King G, Mishra L, Missiuna C. An integrated model of social environment and social context for pediatric rehabilitation. Disabil Rehabil 2016; 38 (12) 1204-1215