Original ArticleThe long-term neurodevelopmental and psychological outcomes of gastroschisis: A cohort study☆,☆☆
Section snippets
Background
Gastroschisis is a relatively common and serious congenital structural anomaly of the abdominal wall and bowel. Most countries report an increasing incidence [1], [2], [3], [4], currently 2.4/10,000 births in Western Australia [5], with survival over 90% [6]. Children born with gastroschisis are frequently exposed to known risk factors for abnormal neuro-psycho-developmental outcome: for example, born to young, single mothers; exposed to maternal vasoactive and illicit drug use, both
Data
All children and adolescents born with gastroschisis in Western Australia between 1992 and 2005 inclusive were identified through the Maternal Fetal Medicine Service Database at King Edward Memorial Hospital for Women (KEMH) and the Princess Margaret Hospital for Children neonatal database (Neobase). These two hospitals are the only tertiary obstetric and children's hospitals in Western Australia. All neonatal gastroschisis cases in Western Australia are managed at Princess Margaret Hospital,
Demographic characteristics
The study cohort consisted of 103 live born children. There were four postnatal deaths resulting in a 96% survival. Gastroschisis was diagnosed antenatally in 93/99 (94%) neonates. All of the study children were born at KEMH and in all of these the gastroschisis was diagnosed antenatally.
We were able to locate 89 of the 99 surviving children and recruit 42 to the study. The median age of the study group was 10 years with a range of 5–17 years. Forty-two children underwent a physical examination
Discussion
This is the only study to date to report a comprehensive assessment of neurodevelopmental and behavioral outcomes in older children and adolescents born with gastroschisis. The intellectual abilities of the children and adolescents in this cohort were substantially similar to the normative population; no child had evidence of intellectual delay (as defined by an IQ < 70); none had evidence of cerebral palsy or hearing loss and only 1 child had a visual defect in the form of amblyopia.
These
Limitations
We recognize that there are some limitations to this study. We could only recruit and evaluate 42% of the cohort. It is possible that children from families of higher socio-economic status would be more likely to participate in the study. However, there was no difference in the Socio-Economic Index for Advantage and Disadvantage between the study and nonstudy group, p = 0.302, according to the Australian Bureau of Statistics' data [35]. Recall bias may mean that those children with ongoing
Conclusions
School aged children and adolescents born with gastroschisis are overall of normal intellect with a small but significant decrease in working memory index. There were no children with cerebral palsy or significant visual or hearing deficits. Of concern however, the children seemed at increased risk for the common pediatric behavioral problems and the parent–child relationship was impaired in many domains. These latter concerns are likely caused by the complex inter relationship between
References (52)
- et al.
Increasing prevalence of gastroschisis in Utah
J Pediatr Surg
(2005) - et al.
The epidemiologic incidence of congenital gastroschisis in Western Australia
Am J Obstet Gynecol
(2003) - et al.
Growth and developmental outcomes of infants with gastroschisis at one year of age: a retrospective study
J Pediatr Surg
(2013) - et al.
Amniotic fluid ferritin as a marker of intestinal damage in gastroschisis: a time course experimental study
J Pediatr Surg
(2007) - et al.
Fetal exposure to an intra-amniotic inflammation and the development of cerebral palsy at the age of three years
Am J Obstet Gynecol
(2000) - et al.
School outcomes of late preterm infants: special needs and challenges for infants born at 32 to 36 weeks gestation
J Pediatr
(2008) - et al.
Developmental progress of newborns undergoing neonatal surgery
J Pediatr Surg
(1990) - et al.
Intellectual development at 3 years of age of children who underwent major neonatal surgery
J Pediatr Surg
(1993) - et al.
Educational attainments in early adolescence of infants who required major neonatal surgery
J Pediatr Surg
(2001) - et al.
Long-term neurodevelopmental outcomes in children born with gastroschisis: the tiebreaker
J Pediatr Surg
(2012)
Early childhood outcomes of infants born with gastroschisis
J Pediatr Surg
The long term physical consequences of gastroschisis
J Pediatr Surg
Psychosocial adjustment and physical growth in children with imperforate anus or abdominal wall defects
J Pediatr Surg
Psychosocial and cognitive consequences of major neonatal surgery
J Pediatr Surg
Interdisciplinary structural follow-up of surgical newborns: a prospective evaluation
J Pediatr Surg
Re: psychosocial and cognitive consequences of major neonatal surgery
J Pediatr Surg
Mother-child interaction and child developmental capacities in toddlers with major birth defects requiring newborn surgery
Early Hum Dev
Congenital anterior abdominal wall defects in England and Wales 1987–93: retrospective analysis of OPCS data
BMJ
Increasing prevalence of gastroschisis in Europe 1980–2002: a phenomenon restricted to younger mothers?
Paediatr Perinat Epidemiol
Changing profile of abdominal wall defects in Japan: results of a national survey
J Pediatr Surg
Recreational drug use: a major risk factor for gastroschisis?
Am J Epidemiol
Rising incidence of gastroschisis in teenage pregnancies
J Matern Fetal Med
Are babies with gastroschisis small for gestational age?
Pediatr Surg Int
Scheduled preterm delivery for gastroschisis improves postoperative outcome
Pediatr Surg Int
Inflammatory response in a rat model of gastroschisis is associated with an increase of NF-kappaB
Braz J Med Biol Res
Biochemical investigations of bowel inflammation in gastroschisis
Pediatr Res
Cited by (35)
Abdominal Wall Defects
2023, Avery's Diseases of the NewbornCongenital anterior abdominal wall defects
2022, Surgery (United Kingdom)Childhood Educational Outcomes of Children Born with Gastroschisis
2022, Journal of PediatricsCitation Excerpt :Studies that specifically assessed the neurodevelopmental outcomes of these infants have included small sample sizes (n <25),16,33 combined different types of abdominal wall defects,17,22,34 assessed noncontemporaneous cohorts,22,34 or limited their analysis to simple gastroschisis.35 In addition, these studies mostly assessed these infants at preschool age (>1 to ≤5 years)33,35-37 or compared their neurodevelopment evaluation with norms rather than control populations.18,20 Some of these studies described similar intelligence20,21 or comparable need for special education services between infants born with gastroschisis and their age peers,38 while others reported higher frequencies of deficits18 or parent-reported developmental delay19 as these children grew older.
Multiple cytokine analysis in gastroschisis: Association with adverse outcomes including fetal brain damage
2021, CytokineCitation Excerpt :Recent studies revealed significant associations between neurodevelopmental impairment and GS. Harris et al. stated that school-age children and adolescents with a history of GS suffer from impaired working memory and have an increased risk for behavioral problems [7]. Other reports demonstrated that children born with GS developed various neurological deficits compared to matched controls [7,21].
Congenital anterior abdominal wall defects
2019, Surgery (United Kingdom)Citation Excerpt :Any abnormalities appear to be correlated with being small-for-gestational age rather than the actual abdominal wall defect. A more recent long-term study of gastroschisis survivors aged 5–17 years old showed overall intellectual abilities again within the normal range.15 A single-centre study from Denmark evaluated the long-term outcomes of a cohort of 40 children born between 1997 and 200316 at a median age of 10.3 years.
Neurodevelopmental outcomes of infants with intestinal failure at 12 and 26 months corrected age
2019, Early Human DevelopmentCitation Excerpt :Although mean ELC scores at 26 months for this sub-set of children remains within the normal range, the decrease between the two time points warrants continued follow up, as it likely indicates increased risk of longer term developmental and learning difficulties. A recent study assessing long-term development and psychological outcomes in children with gastroschisis at a median age of 10 found a decrease in working memory index and behavioural scores [26]. These results highlight the importance of monitoring the neurodevelopment of all children longitudinally regardless of diagnoses or degree of prematurity.
- ☆
Disclosure: The study was funded by a research grant obtained from Channel 7 Telethon, Western Australia.
- ☆☆
The authors declare no conflict of interest, real or perceived by the study sponsor or authors in: (1) study design; (2) the collection, analyfcsis, and interpretation of data; (3) the writing of the report; and (4) the decision to submit the paper for publication. Dr Emma Harris wrote the first draft of the manuscript. No honorarium, grant, or other form of payment was given to anyone to produce the manuscript.