Birth prevalence and survival of exomphalos in england and wales: 2005 to 2011

Birth Defects Res A Clin Mol Teratol. 2014 Sep;100(9):721-5. doi: 10.1002/bdra.23301. Epub 2014 Sep 1.

Abstract

Background: Exomphalos occurs in 2.2 per 10,000 births with 76% of these babies surviving to discharge. The aim of this study was to determine the birth prevalence and survival of babies with this anomaly in England and Wales.

Methods: Six BINOCAR regional congenital anomaly registers in England and Wales (covering 36% of births) between 2005 and 2011 provided cases for this study. Cases included live births, stillbirths (24+ weeks' gestation), late miscarriages (20-23 weeks' gestation), and terminations of pregnancy with fetal anomaly.

Results: The overall birth prevalence was 3.8 (95% confidence interval [CI]: 3.6-4.0) per 10,000 births; 1.4 (1.2-1.6) for isolated cases, 1.2 (1.1-1.4) for cases with multiple anomalies, and 1.2 (1.1-1.4) for cases with chromosomal anomalies. The live birth prevalence was 0.8 (0.7-0.9), 0.5 (0.4-0.6), and 0.1 (0.0-0.1) per 10,000 live births, respectively. Edwards syndrome, congenital heart defects, and nervous system anomalies were the most common anomalies associated with exomphalos. A prenatal diagnosis was made in 83% of isolated, 95% of multiple, and 99% of chromosomal cases. Fifty-five percent of isolated and multiple cases were live born, whereas 85% of cases with chromosomal anomalies resulted in a termination of pregnancy with fetal anomaly. The 1-year survival of live born babies with an isolated exomphalos was 92% compared with 81% in cases with multiple anomalies and 27% in cases with chromosomal anomalies (p < 0.001).

Conclusion: We report a higher birth prevalence than has previously been reported. The proportion of infants surviving with exomphalos remained unchanged over the time period.

Keywords: exomphalos; omphalocele; prenatal diagnosis; prevalence; survival.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Abortion, Eugenic / statistics & numerical data
  • Abortion, Spontaneous / epidemiology*
  • Abortion, Spontaneous / mortality
  • Adolescent
  • Adult
  • Chromosome Aberrations*
  • Chromosomes, Human, Pair 18 / genetics
  • England / epidemiology
  • Female
  • Heart Defects, Congenital / complications
  • Heart Defects, Congenital / diagnosis
  • Heart Defects, Congenital / epidemiology*
  • Heart Defects, Congenital / mortality
  • Hernia, Umbilical / complications
  • Hernia, Umbilical / diagnosis
  • Hernia, Umbilical / epidemiology*
  • Hernia, Umbilical / mortality
  • Humans
  • Live Birth
  • Male
  • Middle Aged
  • Nervous System Malformations / complications
  • Nervous System Malformations / diagnosis
  • Nervous System Malformations / epidemiology*
  • Nervous System Malformations / mortality
  • Pregnancy
  • Prenatal Diagnosis
  • Prevalence
  • Registries
  • Stillbirth
  • Survival Analysis
  • Trisomy / diagnosis*
  • Trisomy / genetics
  • Trisomy 18 Syndrome
  • Wales / epidemiology