{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,28]],"date-time":"2025-12-28T06:18:38Z","timestamp":1766902718676,"version":"3.40.5"},"reference-count":32,"publisher":"Wiley","issue":"12","license":[{"start":{"date-parts":[[2022,5,28]],"date-time":"2022-05-28T00:00:00Z","timestamp":1653696000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100003243","name":"Ministerstvo Zdravotnictv\u00ed Cesk\u00e9 Republiky","doi-asserted-by":"publisher","award":["AZV 17\u201029622A"],"award-info":[{"award-number":["AZV 17\u201029622A"]}],"id":[{"id":"10.13039\/501100003243","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2022,7,15]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Congenital hydrocephalus (CH) comprises a heterogeneous group of birth anomalies with a wide\u2010ranging prevalence across geographic regions and registry type. The aim of the present study was to analyze the early neonatal case fatality rate (CFR) and total birth prevalence of newborns diagnosed with CH.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Data were provided by 25 registries from four continents participating in the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR) on births ascertained between 2000 and 2014. Two CH rates were calculated using a Poisson distribution: early neonatal CFR (death within 7\u2009days) per 100 liveborn CH cases (CFR) and total birth prevalence rate (BPR) per 10,000 births (including live births and stillbirths) (BPR). Heterogeneity between registries was calculated using a meta\u2010analysis approach with random effects. Temporal trends in CFR and BPR within registries were evaluated through Poisson regression modeling.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>A total of 13,112 CH cases among 19,293,280 total births were analyzed. The early neonatal CFR was 5.9 per 100 liveborn cases, 95% confidence interval (CI): 5.4\u20136.8. The CFR among syndromic cases was 2.7 times (95% CI: 2.2\u20133.3) higher than among non\u2010syndromic cases (10.4% [95% CI: 9.3\u201311.7] and 4.4% [95% CI: 3.7\u20135.2], respectively). The total BPR was 6.8 per 10,000 births (95% CI: 6.7\u20136.9). Stratified by elective termination of pregnancy for fetal anomalies (ETOPFA), region and system, higher CFR were observed alongside higher BPR rates. The early neonatal CFR and total BPR did not show temporal variation, with the exception of a CFR decrease in one registry.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Findings of early neonatal CFR and total BPR were highly heterogeneous among registries participating in ICBDSR. Most registries with higher CFR also had higher BPR. Differences were attributable to type of registry (hospital\u2010based vs. population\u2010based), ETOPFA (allowed yes or no) and geographical regions. These findings contribute to the understanding of regional differences of CH occurrence and early neonatal deaths.<\/jats:p><\/jats:sec>","DOI":"10.1002\/bdr2.2045","type":"journal-article","created":{"date-parts":[[2022,5,28]],"date-time":"2022-05-28T09:53:49Z","timestamp":1653731629000},"page":"631-644","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Analysis of early neonatal case fatality rate among newborns with congenital hydrocephalus, a 2000\u20132014 <scp>multi\u2010country registry\u2010based<\/scp> study"],"prefix":"10.1002","volume":"114","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7924-0776","authenticated-orcid":false,"given":"Juan Antonio","family":"Gili","sequence":"first","affiliation":[{"name":"ECLAMC, Centro de Educaci\u00f3n M\u00e9dica e Investigaciones Cl\u00ednicas (CEMIC\u2010CONICET)  Buenos Aires Argentina"},{"name":"Instituto Acad\u00e9mico Pedag\u00f3gico de Ciencias Humanas Universidad Nacional de Villa Mar\u00eda  C\u00f3rdoba Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jorge Santiago","family":"L\u00f3pez\u2010Camelo","sequence":"additional","affiliation":[{"name":"ECLAMC, Centro de Educaci\u00f3n M\u00e9dica e Investigaciones Cl\u00ednicas (CEMIC\u2010CONICET)  Buenos Aires Argentina"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8318-4049","authenticated-orcid":false,"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 University of Arkansas for Medical Sciences  Little Rock Arkansas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marian","family":"Bakker","sequence":"additional","affiliation":[{"name":"Department of Genetics University of Groningen, University Medical Center Groningen, EUROCAT Northern Netherlands  Groningen The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hermien E. K.","family":"de Walle","sequence":"additional","affiliation":[{"name":"Department of Genetics University of Groningen, University Medical Center Groningen, EUROCAT Northern Netherlands  Groningen The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0461-1410","authenticated-orcid":false,"given":"Erin B.","family":"Stallings","sequence":"additional","affiliation":[{"name":"Metro Atlanta Congenital Defects Program (MACDP), Division of Birth Defects and Infant Disorders National Center on Birth Defects and Developmental Disabilities, CDC  Atlanta Georgia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2803-8030","authenticated-orcid":false,"given":"Vijaya","family":"Kancherla","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Emory University Rollins School of Public Health  Atlanta Georgia USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"name":"ECEMC Peripheral Group","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paolo","family":"Contiero","sequence":"additional","affiliation":[{"name":"Lombardy Congenital Anomalies Registry, Cancer Registry Unit, Fondazione IRCCS, Istituto Nazionale Tumori  Milan Italy"}],"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"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9171-7848","authenticated-orcid":false,"given":"Marcia L.","family":"Feldkamp","sequence":"additional","affiliation":[{"name":"Division of Medical Genetics, Department of Pediatrics University of Utah School of Medicine  Salt Lake City Utah USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4326-890X","authenticated-orcid":false,"given":"Amy","family":"Nance","sequence":"additional","affiliation":[{"name":"Utah Birth Defect Network, Bureau of Children with Special Health Care Needs, Division of Family Health and Preparedness Utah Department of Health  Salt Lake City Utah USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Miriam","family":"Gatt","sequence":"additional","affiliation":[{"name":"Malta Congenital Anomalies Registry Directorate for Health Information and Research  Tal\u2010Piet\u00e0 Malta"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Laura","family":"Mart\u00ednez","sequence":"additional","affiliation":[{"name":"Genetics Department, Hospital Universitario Dr. Jos\u00e9 E. 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