{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,22]],"date-time":"2026-10-22T18:49:41Z","timestamp":1792694981309,"version":"4.5.0"},"reference-count":32,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2016,6,14]],"date-time":"2016-06-14T00:00:00Z","timestamp":1465862400000},"content-version":"vor","delay-in-days":13,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2016,6]]},"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Surveillance of congenital anomalies is important to identify potential teratogens. Despite known associations between different anomalies, current surveillance methods examine trends within each subgroup separately. We aimed to evaluate whether hierarchical statistical methods that combine information from several subgroups simultaneously would enhance current surveillance methods using data collected by EUROCAT, a European network of population\u2010based congenital anomaly registries.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Ten\u2010year trends (2003 to 2012) in 18 EUROCAT registries over 11 countries were analyzed for the following groups of anomalies: neural tube defects, congenital heart defects, digestive system, and chromosomal anomalies. Hierarchical Poisson regression models that combined related subgroups together according to EUROCAT's hierarchy of subgroup coding were applied. Results from hierarchical models were compared with those from Poisson models that consider each congenital anomaly separately.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Hierarchical models gave similar results as those obtained when considering each anomaly subgroup in a separate analysis. Hierarchical models that included only around three subgroups showed poor convergence and were generally found to be over\u2010parameterized. Larger sets of anomaly subgroups were found to be too heterogeneous to group together in this way.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>There were no substantial differences between independent analyses of each subgroup and hierarchical models when using the EUROCAT anomaly subgroups. Considering each anomaly separately, therefore, remains an appropriate method for the detection of potential changes in prevalence by surveillance systems. Hierarchical models do, however, remain an interesting alternative method of analysis when considering the risks of specific exposures in relation to the prevalence of congenital anomalies, which could be investigated in other studies. Birth Defects Research (Part A) 106:480\u201310, 2016. \u00a9 2016 Wiley Periodicals, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/bdra.23515","type":"journal-article","created":{"date-parts":[[2016,6,15]],"date-time":"2016-06-15T14:03:39Z","timestamp":1465999419000},"page":"480-488","source":"Crossref","is-referenced-by-count":3,"title":["Use of hierarchical models to analyze European trends in congenital anomaly prevalence"],"prefix":"10.1002","volume":"106","author":[{"given":"Alana","family":"Cavadino","sequence":"first","affiliation":[{"name":"Wolfson Institute of Preventive Medicine Queen Mary University of London  United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Prieto\u2010Merino","sequence":"additional","affiliation":[{"name":"Faculty of Epidemiology and Population Health London School of Hygiene and Tropical Medicine London United Kingdom"},{"name":"Farr Institute of Health Informatics Research University College London  United Kingdom"},{"name":"Catholic University of Murcia (UCAM)  Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie\u2010Claude","family":"Addor","sequence":"additional","affiliation":[{"name":"Service de Genetique Medicale Maternite CHUV Lausanne Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Larraitz","family":"Arriola","sequence":"additional","affiliation":[{"name":"Public Health Division of Gipuzkoa Instituto BIO\u2010Donostia Basque Government CIBER Epidemiolog\u00eda y Salud P\u00fablica \u2010 CIBERESP San Sebatian Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fabrizio","family":"Bianchi","sequence":"additional","affiliation":[{"name":"CNR Institute of Clinical Physiology and Tuscany Registry of Congenital Defects \u201cGabriele Monasterio\u201d Foundation Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth","family":"Draper","sequence":"additional","affiliation":[{"name":"Department of Health Sciences University of Leicester Leicester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ester","family":"Garne","sequence":"additional","affiliation":[{"name":"Paediatric Department Hospital Lillebaelt\u2010Kolding  Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruth","family":"Greenlees","sequence":"additional","affiliation":[{"name":"Institute of Nursing and Health Research Ulster University Newtownabbey United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin","family":"Haeusler","sequence":"additional","affiliation":[{"name":"Medical University of Graz  Austria"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Babak","family":"Khoshnood","sequence":"additional","affiliation":[{"name":"Obstetrical, Perinatal and Pediatric Epidemiology Research Team Center for Biostatistics and Epidemiology INSERM U1153, Maternit\u00e9 de Port\u2010Royal Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jenny","family":"Kurinczuk","sequence":"additional","affiliation":[{"name":"National Perinatal Epidemiology Unit, Nuffield Department of Population Health University of Oxford  United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bob","family":"McDonnell","sequence":"additional","affiliation":[{"name":"Health Service Executive Dublin Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vera","family":"Nelen","sequence":"additional","affiliation":[{"name":"Provincial Institute for Hygiene Antwerp Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mary","family":"O'Mahony","sequence":"additional","affiliation":[{"name":"Department of Public Health Health Service Executive \u2010 South  Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hanitra","family":"Randrianaivo","sequence":"additional","affiliation":[{"name":"Medical Genetics Unit of CHU Sud R\u00e9union Ile de la Reunion  France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judith","family":"Rankin","sequence":"additional","affiliation":[{"name":"Institute of Health &amp; 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