{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T08:08:50Z","timestamp":1780387730303,"version":"3.54.1"},"reference-count":40,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2026,2,20]],"date-time":"2026-02-20T00:00:00Z","timestamp":1771545600000},"content-version":"vor","delay-in-days":19,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,2,1]],"date-time":"2026-02-01T00:00:00Z","timestamp":1769904000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100000030","name":"Centers for Disease Control and Prevention","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000030","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006827","name":"Florida Department of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006827","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100008145","name":"Florida Agency for Health Care Administration","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100008145","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":[[2026,2]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>To estimate sibling recurrence risk for non\u2010chromosomal birth defects using statewide, population\u2010based data.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We analyzed data from the Florida Birth Defects Registry to identify singleton siblings born 2000\u20132019 with any of 43 non\u2010chromosomal birth defects. Multivariable logistic regression was used to estimate adjusted odds ratios (ORs) and 95% confidence intervals (CIs) for the association between birth defect occurrence in the older sibling (exposure) and in the younger sibling (outcome). Two analytic scenarios were considered: (1) any birth defect in the younger sibling, and (2) concordant recurrence, defined as a defect within the same body region. Models adjusted for maternal sociodemographic and perinatal characteristics from linked birth certificate and hospital discharge data.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>The study population included 1,372,831 sibling pairs, of which 29,611 (2.2%) had an older sibling with a birth defect. Among sibling pairs in which the older sibling was affected, 4.8% of younger siblings also had a birth defect. Younger siblings had over twice the odds of having any birth defect if their older sibling was affected compared to those with an unaffected older sibling (adjusted ROR: 2.1, 95% CI: 2.0\u20132.2). Recurrence risk varied by body system, with the highest adjusted odds of concordant recurrence observed for orofacial defects (adjusted ROR: 18.7, 95% CI: 13.9\u201325.1).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Our findings provide contemporary, system\u2010specific estimates of sibling recurrence that may inform family counseling, preconception care, and clinical decision\u2010making.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/bdr2.70034","type":"journal-article","created":{"date-parts":[[2026,2,21]],"date-time":"2026-02-21T02:04:37Z","timestamp":1771639477000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Sibling Recurrence Risk of Non\u2010Chromosomal Birth Defects: A Population\u2010Based Study in Florida, 2000\u20132019"],"prefix":"10.1002","volume":"118","author":[{"given":"Rachel E.","family":"Rutkowski","sequence":"first","affiliation":[{"name":"Department of Epidemiology University of South Florida  Tampa Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean Paul","family":"Tanner","sequence":"additional","affiliation":[{"name":"Department of Epidemiology University of South Florida  Tampa Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3316-2797","authenticated-orcid":false,"given":"Amanda L.","family":"Elmore","sequence":"additional","affiliation":[{"name":"Department of Community Health Sciences and Chiles Center College of Public Health, University of South Florida  Tampa Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3489-401X","authenticated-orcid":false,"given":"Russell S.","family":"Kirby","sequence":"additional","affiliation":[{"name":"Department of Community Health Sciences and Chiles Center College of Public Health, University of South Florida  Tampa Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5874-4155","authenticated-orcid":false,"given":"A. J.","family":"Agopian","sequence":"additional","affiliation":[{"name":"Department of Epidemiology University of Texas Health Science Center (UTHealth) School of Public Health  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0077-6023","authenticated-orcid":false,"given":"Jason L.","family":"Salemi","sequence":"additional","affiliation":[{"name":"Department of Epidemiology University of South Florida  Tampa Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,2,20]]},"reference":[{"key":"e_1_2_13_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.1387"},{"key":"e_1_2_13_3_1","doi-asserted-by":"publisher","DOI":"10.1093\/oxfordjournals.aje.a010058"},{"key":"e_1_2_13_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.38237"},{"key":"e_1_2_13_5_1","unstructured":"Centers for Disease Control and Prevention.2023a. \u201cCommit to Healthy Choices to Help Prevent Birth Defects.\u201dhttps:\/\/www.cdc.gov\/birth\u2010defects\/prevention\/?CDC_AAref_Val=https:\/\/www.cdc.gov\/ncbddd\/birthdefects\/prevention.html."},{"key":"e_1_2_13_6_1","unstructured":"Centers for Disease Control and Prevention.2023b. \u201cData and Statistics on Birth Defects.\u201dhttps:\/\/www.cdc.gov\/birth\u2010defects\/data\u2010research\/facts\u2010stats\/?CDC_AAref_Val=https:\/\/www.cdc.gov\/ncbddd\/birthdefects\/data.html."},{"key":"e_1_2_13_7_1","unstructured":"Centers for Disease Control and Prevention.2023c. \u201cFacts About Cleft Lip and Cleft Palate.\u201dhttps:\/\/www.cdc.gov\/birth\u2010defects\/about\/cleft\u2010lip\u2010cleft\u2010palate.html?CDC_AAref_Val=https:\/\/www.cdc.gov\/ncbddd\/birthdefects\/cleftlip.html."},{"issue":"1","key":"e_1_2_13_8_1","first-page":"182","article-title":"Familial Recurrence\u2010Pattern Analysis of Nonsyndromic Isolated Cleft Palate\u2014A Danish Registry Study","volume":"58","author":"Christensen K.","year":"1996","journal-title":"American Journal of Human Genetics"},{"key":"e_1_2_13_9_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199507203330305"},{"key":"e_1_2_13_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20619"},{"key":"e_1_2_13_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23295"},{"key":"e_1_2_13_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33099"},{"key":"e_1_2_13_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23576"},{"key":"e_1_2_13_14_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12916\u2010017\u20100789\u20105"},{"key":"e_1_2_13_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33608"},{"key":"e_1_2_13_16_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2009.069385"},{"key":"e_1_2_13_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.annepidem.2017.10.010"},{"key":"e_1_2_13_18_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199407073310101"},{"key":"e_1_2_13_19_1","doi-asserted-by":"publisher","DOI":"10.18632\/oncotarget.19890"},{"issue":"9","key":"e_1_2_13_20_1","first-page":"1","article-title":"Infant Mortality Statistics From the 2013 Period Linked Birth\/Infant Death Data Set","volume":"64","author":"Matthews T. J.","year":"2015","journal-title":"National Vital Statistics Reports"},{"key":"e_1_2_13_21_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365\u20103016.11.s1.3.x"},{"key":"e_1_2_13_22_1","unstructured":"National Institutes of Health.2017. \u201cWhat Causes Birth Defects.\u201dhttps:\/\/www.nichd.nih.gov\/health\/topics\/birthdefects\/conditioninfo\/causes."},{"key":"e_1_2_13_23_1","unstructured":"National Institutes of Health.2020. \u201cBirth Defects.\u201dhttps:\/\/www.nichd.nih.gov\/health\/topics\/birthdefects."},{"key":"e_1_2_13_24_1","doi-asserted-by":"publisher","DOI":"10.1093\/aje\/kwp087"},{"key":"e_1_2_13_25_1","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.109.857987"},{"issue":"3","key":"e_1_2_13_26_1","first-page":"91","article-title":"Improving the Completeness of Ascertainment in Florida's Birth Defects Surveillance Program: The Impact of Adding Infant Death and Emergency Department Data","volume":"42","author":"Rutkowski R. E.","year":"2015","journal-title":"Journal of Registry Management"},{"issue":"2","key":"e_1_2_13_27_1","first-page":"48","article-title":"Evaluating Difficult Decisions in Public Health Surveillance: Striking the Right Balance Between Timeliness and Completeness","volume":"42","author":"Salemi J. L.","year":"2015","journal-title":"Journal of Registry Management"},{"issue":"1","key":"e_1_2_13_28_1","first-page":"14","article-title":"Creation and Evaluation of a Multi\u2010Layered Maternal and Child Health Database for Comparative Effectiveness Research","volume":"40","author":"Salemi J. L.","year":"2013","journal-title":"Journal of Registry Management"},{"issue":"1","key":"e_1_2_13_29_1","first-page":"30","article-title":"The Relative Contribution of Data Sources to a Birth Defects Registry Utilizing Passive Multisource Ascertainment Methods: Does a Smaller Birth Defects Case Ascertainment Net Lead to Overall or Disproportionate Loss?","volume":"38","author":"Salemi J. 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Average Number of People Per Family Household With Own Children Under 18 by Race and Hispanic Origin Marital Status Age and Education of Householder: 2020.\u201dhttps:\/\/www.census.gov\/data\/tables\/2020\/demo\/families\/cps\u20102020.html."},{"key":"e_1_2_13_36_1","unstructured":"United States Census Bureau.2021. \u201cHispanic or Latino Origin by Race. 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