Elsevier

Early Human Development

Volume 103, December 2016, Pages 209-218
Early Human Development

Outcome of isolated gastroschisis; an international study, systematic review and meta-analysis

https://doi.org/10.1016/j.earlhumdev.2016.10.002Get rights and content

Abstract

Objective

To determine outcome of children born with isolated gastroschisis (no extra-gastrointestinal congenital abnormalities).

Study design

International cohort study and meta-analysis. Primary outcome: time to full enteral feeding (TFEF); secondary outcomes: Duration of mechanical ventilation, length of stay (LOS), mortality and differences in outcome between simple and complex gastroschisis (complex; born with bowel atresia, volvulus, perforation or necrosis).

To compare the cohort study results with literature three databases were searched. Studies were eligible for inclusion if cases were born in developed countries with isolated gastroschisis after 1990, number of cases > 20 and TFEF was reported.

Results

The cohort study included 204 liveborn cases of isolated gastroschisis. The TFEF, median duration of ventilation and LOS was, 26 days (range 6–515), 2 days (range 0–90) and 33 days (range 11–515), respectively. Overall mortality was 10.8%. TFEF and LOS were significantly longer (P < 0.0001) and mortality was fourfold higher in the complex group. Seventeen studies, amongst the current study, were included for further meta-analysis comprising a total of 1652 patients. Mean TFEF was 35.3 ± 4.4 days, length of ventilation was 5.5 ± 2.0 days, LOS was 46.4 ± 5.2 days and mortality risk was 0.06 [0.04–0.07 95%CI].

Outcome of simple and complex gastroschisis was described in five studies. TFEF, ventilation time, LOS were significant longer and mortality rate was 3.64 [1.95–6.83 95%CI] times higher in complex cases.

Conclusions

These results give a good indication of the expected TFEF, ventilation time and LOS and mortality risk in children born with isolated gastroschisis, although ranges remain wide. This study shows the importance of dividing gastroschisis into simple and complex for the prediction of outcome.

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:

  • Gastroschisis is a structural anomaly with an increased perinatal risk of morbidity and mortality

  • Bowel damage at birth influences outcome

What this study adds:

  • First systematic review on isolated gastroschisis outcome; time to full enteral feedings and length of mechanical ventilation, hospital stay and mortality

  • 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.

References (52)

  • S.Y. Segel et al.

    Fetal abdominal wall defects and mode of delivery: a systematic review

    Obstet. Gynecol.

    (2001 Nov)
  • M.E. Abdel-Latif et al.

    Australian and New Zealand neonatal network. Mode of delivery and neonatal survival of infants with gastroschisis in Australia and New Zealand

    J. Pediatr. Surg.

    (2008 Sep)
  • R.M. Sydorak et al.

    Gastroschisis: small hole, big cost

    J. Pediatr. Surg.

    (2002 Dec)
  • M.A. Arnold et al.

    Risk stratification of 4344 patients with gastroschisis into simple and complex categories

    J. Pediatr. Surg.

    (2007 Sep)
  • O. Ergun et al.

    The timing of delivery of infants with gastroschisis influences outcome

    J. Pediatr. Surg.

    (2005 Feb)
  • G.H. Visser et al.

    New dutch reference curves for birthweight by gestational age

    Early Hum. Dev.

    (2009 Dec)
  • J. Huang et al.

    Benefits of term delivery in infants with antenatally diagnosed gastroschisis

    Obstet. Gynecol.

    (2002 Oct)
  • E.Y. Yang et al.

    Spontaneous onset of labor, not route of delivery, is associated with prolonged length of stay in babies with gastroschisis

    J. Pediatr. Surg.

    (2014 Dec)
  • A.P. South et al.

    Meta-analysis of the prevalence of intrauterine fetal demise in gastroschisis

    Am. J. Obstet. Gynecol.

    (2013)
  • J. Rankin et al.

    Congenital anterior abdominal wall defects in the north of England, 1986-1996: occurrence and outcome

    Prenat. Diagn.

    (1999 Jul)
  • E. Calzolari et al.

    Omphalocele and gastroschisis in europe: a survey of 3 million births 1980-1990. EUROCAT working group

    Am. J. Med. Genet.

    (1995 Aug 28)
  • P. Mastroiacovo et al.

    Gastroschisis and associated defects: an international study

    Am. J. Med. Genet. A

    (2007 Apr 1)
  • C. Stoll et al.

    Omphalocele and gastroschisis and associated malformations

    Am. J. Med. Genet. A

    (2008 May 15)
  • J. Garvin et al.

    Gastroschisis complicated by septo-optic dysplasia: two distinct anomalies with a common origin

    AJP Rep.

    (2016)
  • F. Abdullah et al.

    Gastroschisis in the United States 1988-2003: analysis and risk categorization of 4344 patients

    J. Perinatol.

    (2007 Jan)
  • D. Moher et al.

    PRISMA group. Preferred reporting items for systematic reviews and meta-analyses: The PRISMA statement. BMJ

    (2009)
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