Original Article
The long-term neurodevelopmental and psychological outcomes of gastroschisis: A cohort study,☆☆

https://doi.org/10.1016/j.jpedsurg.2015.08.062Get rights and content

Abstract

Objectives

Previous gastroschisis specific neurodevelopmental studies have focused on the first 3 years of life. The aim of this study was to assess the intellectual, behavioral and neurological outcomes of older children and adolescents born with gastroschisis.

Study design

Of 99 gastroschisis survivors born in Western Australia, 1992 to 2005, and who were at least 5 years old, 42 agreed to take part in this study. The study assessed: intellectual ability, with age appropriate Wechsler intelligence scales; neurological status; hearing; vision; behavioral status with the Strengths and Difficulties Questionnaire (SDQ); and parenting style with the Parenting Relationship Questionnaire (PRQ). All results were compared to normative means.

Results

Median age at follow-up was 10 years (range 5–17). No child had evidence of cerebral palsy or hearing loss; 1 child had amblyopia. Psychometric tests were completed in 39 children: mean full scale IQ was 98.2 (standard deviation [SD] 10.7); the working memory index was the only subscale to show a significant decrease from the normative mean (mean 95.5, SD 12.4, p = 0.038). The mean SDQ behavioral scores were significantly lower for 3 of 5 domains and the Total Difficulties score. PRQ scores were significantly abnormal for 4 of 7 domains: Communication, Discipline, Satisfaction with School and Relational Frustration.

Conclusions

Overall intellectual abilities were within a normal range. The decrease in working memory index and the behavioral and parenting relationship impairments could be an effect of perinatal factors, gastroschisis management and complications or the complexity of the socio-economic environment.

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

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    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.

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