{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,1]],"date-time":"2026-05-01T15:11:28Z","timestamp":1777648288676,"version":"3.51.4"},"reference-count":27,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2019,10,21]],"date-time":"2019-10-21T00:00:00Z","timestamp":1571616000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Paediatric Perinatal Epid"],"published-print":{"date-parts":[[2019,11]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Medical advancements have resulted in better survival and life expectancy among those with spina bifida, but a significantly increased risk of perinatal and postnatal mortality for individuals with spina bifida remains.<\/jats:p><\/jats:sec><jats:sec><jats:title>Objectives<\/jats:title><jats:p>To examine stillbirth and infant and child mortality among those affected by spina bifida using data from multiple countries.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We conducted an observational study, using data from 24 population\u2010 and hospital\u2010based surveillance registries in 18 countries contributing as members of the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR). Cases of spina bifida that resulted in livebirths or stillbirths from 20\u00a0weeks' gestation or elective termination of pregnancy for fetal anomaly (ETOPFA) were included. Among liveborn spina bifida cases, we calculated mortality at different ages as number of deaths among liveborn cases divided by total number of liveborn cases with spina bifida. As a secondary outcome measure, we estimated the prevalence of spina bifida per 10\u00a0000 total births. The 95% confidence interval for the prevalence estimate was estimated using the Poisson approximation of binomial distribution.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Between years 2001 and 2012, the overall first\u2010week mortality proportion was 6.9% (95% CI 6.3, 7.7) and was lower in programmes operating in countries with policies that allowed ETOPFA compared with their counterparts (5.9% vs. 8.4%). The majority of first\u2010week mortality occurred on the first day of life. In programmes where information on long\u2010term mortality was available through linkage to administrative databases, survival at 5\u00a0years of age was 90%\u201096% in Europe, and 86%\u201096% in North America.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Our multi\u2010country study showed a high proportion of stillbirth and infant and child deaths among those with spina bifida. Effective folic acid interventions could prevent many cases of spina bifida, thereby preventing associated childhood morbidity and mortality.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ppe.12589","type":"journal-article","created":{"date-parts":[[2019,10,22]],"date-time":"2019-10-22T02:14:02Z","timestamp":1571710442000},"page":"436-448","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":36,"title":["Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry\u2010Based Study, 2001\u20102012"],"prefix":"10.1111","volume":"33","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5098-6400","authenticated-orcid":false,"given":"Marian K.","family":"Bakker","sequence":"first","affiliation":[{"name":"University of Groningen University Medical Center Groningen Department of Genetics Eurocat Northern Netherlands Groningen The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vijaya","family":"Kancherla","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Emory University Rollins School of Public Health Atlanta GA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mark A.","family":"Canfield","sequence":"additional","affiliation":[{"name":"Birth Defects Epidemiology and Surveillance Branch Texas Department of State Health Services Austin TX US"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eva","family":"Bermejo\u2010Sanchez","sequence":"additional","affiliation":[{"name":"ECEMC (Spanish Collaborative Study of Congenital Malformations) CIAC Instituto de Investigaci\u00f3n de Enfermedades Raras (IIER) Instituto de Salud Carlos III Madrid Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Janet D.","family":"Cragan","sequence":"additional","affiliation":[{"name":"Division of Congenital and Developmental Disorders National Center on Birth Defects and Development Disabilities Centers for Disease Control Atlanta GA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Saeed","family":"Dastgiri","sequence":"additional","affiliation":[{"name":"Health Services Management Research Centre Tabriz University of Medical Sciences Tabriz Iran"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hermien E. K.","family":"De Walle","sequence":"additional","affiliation":[{"name":"University of Groningen University Medical Center Groningen Department of Genetics Eurocat Northern Netherlands Groningen The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marcia L.","family":"Feldkamp","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Utah School of Medicine and the Utah Birth Defect Network Salt Lake City UT USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Boris","family":"Groisman","sequence":"additional","affiliation":[{"name":"National Network of Congenital Anomalies of Argentina (RENAC) National Center of Medical Genetics National Administration of Laboratories and Health Institutes (ANLIS) National Ministry of Health Buenos Aires Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Miriam","family":"Gatt","sequence":"additional","affiliation":[{"name":"Malta Congenital Anomalies Registry Directorate for Health Information and Research Valetta Malta"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paula","family":"Hurtado\u2010Villa","sequence":"additional","affiliation":[{"name":"Department of Basic Sciences of Health School of Health Pontificia Universidad Javeriana Cali Cali Colombia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Karin","family":"Kallen","sequence":"additional","affiliation":[{"name":"National Board of Health and Welfare and University of Lund Stockholm Sweden"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daniella","family":"Landau","sequence":"additional","affiliation":[{"name":"Department of Neonatology Soroka Medical Center Beer\u2010Sheva Israel"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nathalie","family":"Lelong","sequence":"additional","affiliation":[{"name":"Inserm UMR 1153 Obstetrical Perinatal and Pediatric Epidemiology Research Team (Epop\u00e9) Center for Epidemiology and Statistics Sorbonne Paris Cit\u00e9 DHU Risks in Pregnancy Paris Descartes University Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jorge S.","family":"Lopez Camelo","sequence":"additional","affiliation":[{"name":"ECLAMC Center for Medical Education and Clinical Research (CEMIC\u2010CONICET) Buenos Aires Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Laura","family":"Mart\u00ednez","sequence":"additional","affiliation":[{"name":"Genetics Department Hospital Universitario Dr Jose E. Gonzalez Universidad Auton\u00f3ma de Nuevo Le\u00f3n San Nicol\u00e1s de los Garza Mexico"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Margery","family":"Morgan","sequence":"additional","affiliation":[{"name":"CARIS, The Congenital Anomaly Register for Wales Singleton Hospital Swansea UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Osvaldo M.","family":"Mutchinick","sequence":"additional","affiliation":[{"name":"RYVEMCE Department of Genetics Instituto Nacional de Ciencias M\u00e9dicas y Nutrici\u00f3n Salvador Zubir\u00e1n Mexico City Mexico"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wendy N.","family":"Nembhard","sequence":"additional","affiliation":[{"name":"Department of Epidemiology, Arkansas Center for Birth Defects Research and Prevention and Arkansas Reproductive Health Monitoring System Fay Boozman College of Public Health University of Arkansas for Medical Sciences Little Rock AR USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anna","family":"Pierini","sequence":"additional","affiliation":[{"name":"Institute of Clinical Physiology National Research Council and Fondazione Toscana Gabriele Monasterio Tuscany Registry of Congenital Defects Pisa Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anke","family":"Rissmann","sequence":"additional","affiliation":[{"name":"Malformation Monitoring Centre Saxony\u2010Anhalt Medical Faculty Otto\u2010von\u2010Guericke University Magdeburg Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Antonin","family":"Sipek","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics Thomayer Hospital Prague Czech Republic"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Elena","family":"Szabova","sequence":"additional","affiliation":[{"name":"Slovak Teratologic Information Centre (FPH) Slovak Medical University Bratislava Slovak Republic"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Giovanna","family":"Tagliabue","sequence":"additional","affiliation":[{"name":"Lombardy Congenital Anomalies Registry Cancer Registry Unit Fondazione IRCCS Istituto Nazionale tumori Milan Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wladimir","family":"Wertelecki","sequence":"additional","affiliation":[{"name":"Omni\u2010Net for Children International Charitable Fund Rivne Rivne Ukraine"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ignacio","family":"Zarante","sequence":"additional","affiliation":[{"name":"Human Genetics Institute Pontificia Universidad Javeriana Bogot\u00e1 Colombia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pierpaolo","family":"Mastroiacovo","sequence":"additional","affiliation":[{"name":"International Center on Birth Defects International Clearinghouse for Birth Defects Surveillance and Research Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2019,10,21]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199911113412006"},{"key":"e_1_2_8_3_1","volume-title":"Global Report on Birth Defects: The Hidden Toll of Dying and Disabled Children","author":"Christianson AL","year":"2006"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.2105\/AJPH.2015.302902"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0151586"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/nyas.13548"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(80)90886-7"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/0140-6736(91)90133-A"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199212243272602"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.annepidem.2009.01.016"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.2016.19438"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(74)92606-3"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20498"},{"key":"e_1_2_8_14_1","first-page":"213","article-title":"Anencephalus and spina bifida in Belfast (1964\u20101968)","volume":"42","author":"Elwood JH","year":"1973","journal-title":"Ulster Med J"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1515\/JPM.2005.062"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2009.03543.x"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.22858"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2012.05.040"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23259"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-3016.1994.tb00467.x"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-3016.2001.00371.x"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00381-017-3685-6"},{"key":"e_1_2_8_23_1","first-page":"270","article-title":"Meningomyelocele: management in utero and post natum","volume":"181","author":"Shurtleff DB","year":"1994","journal-title":"Ciba Found Symp"},{"key":"e_1_2_8_24_1","first-page":"568","article-title":"The median survival time in open spina bifida","volume":"39","author":"Hunt GM","year":"1997","journal-title":"Dev Med Child Neurol"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2005-1364"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1159\/000056005"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20711"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1002\/uog.12546"}],"container-title":["Paediatric and Perinatal Epidemiology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/ppe.12589","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/ppe.12589","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/ppe.12589","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,9]],"date-time":"2023-09-09T00:15:25Z","timestamp":1694218525000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/ppe.12589"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,10,21]]},"references-count":27,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2019,11]]}},"alternative-id":["10.1111\/ppe.12589"],"URL":"https:\/\/doi.org\/10.1111\/ppe.12589","archive":["Portico"],"relation":{},"ISSN":["0269-5022","1365-3016"],"issn-type":[{"value":"0269-5022","type":"print"},{"value":"1365-3016","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,10,21]]},"assertion":[{"value":"2019-05-09","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2019-08-24","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2019-10-21","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}