Outcome of isolated gastroschisis; an international study, systematic review and meta-analysis
Introduction
Nowadays, gastroschisis is nearly always diagnosed prenatally during routine first and second trimester ultrasound examinations. However, even with early prenatal diagnosis, a recent meta-analysis has shown that intra-uterine fetal death is still 7-fold higher (4.48%) compared to the general population (0.62%) [1]. Neonatal survival and quality of life of children born with gastroschisis are often expressed as excellent, however, numbers differ widely between studies. This quantitative wide range might be the result of different treatment strategies, or caused by the fact that most studies have included gastroschisis cases with additional extra-intestinal congenital abnormalities [2]. The incidence of associated anomalies in gastroschisis varies from 5 to over 20% between studies [3], [4]. Reported associations include cardiac abnormalities and increased prevalence of central nervous system anomalies (amyoplasia), and limb and kidney anomalies [5], [6], [7] and may influence the prognosis of the child with gastroschisis significantly [8].
With this study we aimed to determine the outcome of children born with isolated gastroschisis in order to give the prognosis of solely the entity gastroschisis in a cohort of 204 cases and to systematically review the literature to compare our findings with studies describing isolated gastroschisis cases born in other Western countries. Our primary objective was to investigate the time to full enteral feedings (TFEF) in isolated cases of gastroschisis, since this reflects the condition of the child and its bowel, and secondarily to investigate length of mechanical ventilation, length of hospital stay (LOS) and mortality. In addition, we investigated the difference between simple and complex gastroschisis (additional atresia, volvulus, perforation or necrosis of the bowel at birth) [9] on outcome measurements. It was our goal to provide future parents and clinicians with more quantitative data regarding outcome of their child with isolated gastroschisis.
Section snippets
Retrospective study
We conducted a cohort study of all live born gastroschisis cases, treated between January 2002 and January 2010 in six university hospitals in the Netherlands (‘Netherlands’) (Academic Medical Center Amsterdam, Maastricht University Medical Center +, Radboud University Medical Center Nijmegen, VU University Medical Center Amsterdam, University Medical Center Groningen, University Medical Center Utrecht) or born and treated at the Hospital das Clínicas da Faculdade de Medicina da Universidade de
Gastroschisis cohort of the Netherlands and Brazil
A total of 204 live born cases of isolated gastroschisis were identified. The characteristics of the cases treated in the Netherlands and Brazil are presented and compared in Table 1. The gestational age and subsequent birth weight of cases treated in Brazil was significantly lower compared to cases treated at the Netherlands. However, the percentage of cases born with a birth weight < 10 percentile was comparable between both groups (31.4% versus 38.2%, P = 0.38). As expected, the CS rate was
Discussion
This study is the first meta-analysis on outcome of isolated gastroschisis cases, providing quantitative outcome measures which can be used for counseling future parents. The mortality, average TFEF, length of ventilation, and LOS of the current study were in line with the studies from the systematic review and can be used for the counseling of parents. This study also confirms that children with a simple gastroschisis have a better outcome compared to children with a complex gastroschisis.
The
Conclusion
This international cohort study and systematic review focused on isolated gastroschisis outcome regarding, TFEF, ventilation duration, LOS and mortality. Although there was a wide range in outcome, these quantitative data may provide future parents and clinicians a better indication of the expected prognosis. This study also shows the importance of classification of cases into simple and complex cases for the prediction of outcome.
What's known on this subject:
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Gastroschisis is a structural anomaly with an increased perinatal risk of morbidity and mortality
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Bowel damage at birth influences outcome
What this study adds:
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First systematic review on isolated gastroschisis outcome; time to full enteral feedings and length of mechanical ventilation, hospital stay and mortality
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Outcome depends on the presence of simple as compared to complex gastroschisis
Financial disclosure statement
Nothing to disclose.
Conflict of interest statement for all authors
No conflicts.
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2022, Jornal de PediatriaCitation Excerpt :While Raymond et al.8 found a 5% mortality in an American multicenter study, mortality in the northern region of Brazil was 51.2% according to Bilibio et al.17 and 11.4% in the southeast region of Brazil.18 In Europe, in a meta-analysis published in 2016,19 mortality in the Netherlands was 8.8%. These differences can be also be explained by lower adequate prenatal rates of pregnant women, the presence of more cases of complex gastroschisis, and infections.
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