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Neurodevelopmental outcome of patients with congenital gastrointestinal malformations: a systematic review and
Daniëlle Roorda1,2, Marsh Königs2, Laurens Eeftinck Schattenkerk3
1Department of Pediatric Surgery, Amsterdam Reproduction and Development Research Institute, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam and Free University Amsterdam, Amsterdam, The Netherlands d.roorda@amsterdamumc.nl.
Insights
Children with congenital gastrointestinal malformations often experience neurodevelopmental impairments, including cognitive, motor, and language deficits. Early screening for neurodevelopmental outcomes is recommended for these vulnerable children.
Area of Science:
- Pediatric Surgery
- Neurodevelopmental Pediatrics
- Gastroenterology
Background:
- Congenital gastrointestinal malformations pose risks to developing brains.
- Perioperative factors, anesthesia, and inflammation can impact neurodevelopment.
- Existing evidence on neurodevelopmental outcomes requires aggregation.
Purpose of the Study:
- To systematically review and meta-analyze neurodevelopmental outcomes in children with congenital gastrointestinal malformations.
- To quantify the extent of cognitive, motor, and language impairments.
- To identify factors influencing neurodevelopmental outcomes in this population.
Main Methods:
- Comprehensive literature search of PubMed, Embase, and Web of Science databases.
- Inclusion of 47 studies representing 62 cohorts and 2312 patients.
- Random-effects meta-analysis of standardized mean differences (Cohen's d) compared to normative data or control groups.
Main Results:
- Children exhibited significant cognitive (d=-0.435), motor (d=-0.610), and language (d=-0.670) impairments.
- Short bowel syndrome was associated with poorer motor outcomes.
- Neurodevelopmental outcomes correlated with surgery number and hospital stay duration.
Conclusions:
- Children with congenital gastrointestinal malformations demonstrate neurodevelopmental deficits.
- Routine neurodevelopmental screening is crucial during follow-up care.
- Further research into specific interventions is warranted.
Aim:
Children with congenital gastrointestinal malformations may be at risk of neurodevelopmental impairment due to challenges to the developing brain, including perioperative haemodynamic changes, exposure to anaesthetics and postoperative inflammatory influences. This study aggregates existing evidence on neurodevelopmental outcome in these patients using meta-analysis.
Method:
PubMed, Embase and Web of Science were searched for peer-reviewed articles published until October 2019. Out of the 5316 unique articles that were identified, 47 studies met the inclusion criteria and were included. Standardised mean differences (Cohen's d) between cognitive, motor and language outcome of patients with congenital gastrointestinal malformations and normative data (39 studies) or the studies' control group (8 studies) were aggregated across studies using random-effects meta-analysis. The value of (clinical) moderators was studied using meta-regression and diagnostic subgroups were compared.
Results:
The 47 included studies encompassed 62 cohorts, representing 2312 patients. Children with congenital gastrointestinal malformations had small-sized cognitive impairment (d=-0.435, p<0.001; 95% CI -0.567 to -0.302), medium-sized motor impairment (d=-0.610, p<0.001; 95% CI -0.769 to -0.451) and medium-sized language impairment (d=-0.670, p<0.001; 95% CI -0.914 to -0.425). Patients with short bowel syndrome had worse motor outcome. Neurodevelopmental outcome was related to the number of surgeries and length of total hospital stay, while no relations were observed with gestational age, birth weight, age and sex.
Interpretation:
This study shows that children with congenital gastrointestinal malformations exhibit impairments in neurodevelopmental outcome, highlighting the need for routine screening of neurodevelopment during follow-up.
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