{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,6]],"date-time":"2026-09-06T05:06:31Z","timestamp":1788671191290,"version":"build-2803163510"},"reference-count":23,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2014,9,1]],"date-time":"2014-09-01T00:00:00Z","timestamp":1409529600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2014,9]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>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.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>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\u201323 weeks' gestation), and terminations of pregnancy with fetal anomaly.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      The overall birth prevalence was 3.8 (95% confidence interval [CI]: 3.6\u20134.0) per 10,000 births; 1.4 (1.2\u20131.6) for isolated cases, 1.2 (1.1\u20131.4) for cases with multiple anomalies, and 1.2 (1.1\u20131.4) for cases with chromosomal anomalies. The live birth prevalence was 0.8 (0.7\u20130.9), 0.5 (0.4\u20130.6), and 0.1 (0.0\u20130.1) 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\u2010five 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\u2010year 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 (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.001).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>We report a higher birth prevalence than has previously been reported. The proportion of infants surviving with exomphalos remained unchanged over the time period. Birth Defects Research (Part A) 100:721\u2013725, 2014. \u00a9 2014 Wiley Periodicals, Inc.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/bdra.23301","type":"journal-article","created":{"date-parts":[[2014,9,1]],"date-time":"2014-09-01T09:38:37Z","timestamp":1409564317000},"page":"721-725","source":"Crossref","is-referenced-by-count":41,"title":["Birth prevalence and survival of exomphalos in england and wales: 2005 to 2011"],"prefix":"10.1002","volume":"100","author":[{"given":"Anna","family":"Springett","sequence":"first","affiliation":[{"name":"Centre for Environmental and Preventive Medicine Queen Mary University of London  London"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth S.","family":"Draper","sequence":"additional","affiliation":[{"name":"Department of Health Sciences University of Leicester Leicester"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judith","family":"Rankin","sequence":"additional","affiliation":[{"name":"Institute of Health &amp; Society Newcastle University Newcastle Upon Tyne"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Catherine","family":"Rounding","sequence":"additional","affiliation":[{"name":"National Perinatal Epidemiology Unit University of Oxford Oxford"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Tucker","sequence":"additional","affiliation":[{"name":"Public Health Wales Wales"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sylvia","family":"Stoianova","sequence":"additional","affiliation":[{"name":"Institute of Child Life and Health, St Michael's Hospital Bristol"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Diana","family":"Wellesley","sequence":"additional","affiliation":[{"name":"Faculty of Medicine University of Southampton and Wessex Clinical Genetics Service Southampton"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joan K.","family":"Morris","sequence":"additional","affiliation":[{"name":"Centre for Environmental and Preventive Medicine Queen Mary University of London  London"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2014,9]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33000"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.0960-7692.2001.00534.x"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1148\/radiol.2321030795"},{"key":"e_1_2_6_5_1","first-page":"F209","volume-title":"Arch Dis Child Fetal Neonatal Ed","author":"Boyd PA","year":"1998"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-3016.1998.00090.x"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320580218"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20294"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0002-9610(96)00401-1"},{"key":"e_1_2_6_10_1","unstructured":"EUROCAT. EUROCAT Guide 1.3 and reference documents. September 2005. Available at:http:\/\/www.eurocat\u2010network.eu\/content\/EUROCAT\u2010Guide\u20101.3.pdf. Accessed Month day year."},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3468(96)90491-1"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-9926(199909)60:3<117::AID-TERA5>3.0.CO;2-G"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2005.11.013"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajog.2003.06.007"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1093\/aje\/kwh051"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2008.08.050"},{"key":"e_1_2_6_17_1","first-page":"236","article-title":"Changing trend in congenital abdominal wall defects in eastern region of Ireland","volume":"95","author":"McDonnell R","year":"2002","journal-title":"Ir Med J"},{"key":"e_1_2_6_18_1","volume-title":"Statistical bulletin: live births in England and Wales by characteristics of mother 1","author":"Office for National Statistics (ONS)","year":"2011"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00383-009-2357-3"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-0223(199907)19:7<662::AID-PD607>3.0.CO;2-C"},{"key":"e_1_2_6_21_1","volume-title":"Congenital anomaly statistics 2011","author":"Springett A","year":"2013"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3468(96)90146-3"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.313.7062.903"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(09)61922-X"}],"container-title":["Birth Defects Research Part A: Clinical and Molecular Teratology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fbdra.23301","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/bdra.23301","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,6]],"date-time":"2023-10-06T03:40:37Z","timestamp":1696563637000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/bdra.23301"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,9]]},"references-count":23,"journal-issue":{"issue":"9","published-print":{"date-parts":[[2014,9]]}},"alternative-id":["10.1002\/bdra.23301"],"URL":"https:\/\/doi.org\/10.1002\/bdra.23301","archive":["Portico"],"relation":{},"ISSN":["1542-0752","1542-0760"],"issn-type":[{"value":"1542-0752","type":"print"},{"value":"1542-0760","type":"electronic"}],"subject":[],"published":{"date-parts":[[2014,9]]}}}