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Interest in hydroxychloroquine has increased as it is being examined as a potential treatment and prophylaxis for coronavirus disease 2019. Data on the risks of specific birth defects associated with hydroxychloroquine use are sparse.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Using data from two case\u2013control studies (National Birth Defects Prevention Study and Slone Epidemiology Center Birth Defects Study), we described women who reported hydroxychloroquine use in pregnancy and the presence of specific major birth defects in their offspring. Cases had at least one major birth defect and controls were live\u2010born healthy infants. Women self\u2010reported medication use information in the few months before pregnancy through delivery.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>In total, 0.06% (19\/31,468) of case and 0.04% (5\/11,614) of control mothers in National Birth Defects Prevention Study, and 0.04% (11\/29,838) of case and 0.05% (7\/12,868) of control mothers in Birth Defects Study reported hydroxychloroquine use. Hydroxychloroquine users had complicated medical histories and frequent medication use for a variety of conditions. The observed birth defects among women taking hydroxychloroquine were varied and included nine oral cleft cases; the elevated observed:expected ratios for specific oral cleft phenotypes and for oral clefts overall had 95% confidence intervals that included 1.0.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>While teratogens typically produce a specific pattern of birth defects, the observed birth defects among the hydroxychloroquine\u2010exposed women did not present a clear pattern, suggesting no meaningful evidence for the risk of specific birth defects. The number of exposed cases is small; results should be interpreted cautiously.<\/jats:p><\/jats:sec>","DOI":"10.1002\/bdr2.1943","type":"journal-article","created":{"date-parts":[[2021,7,23]],"date-time":"2021-07-23T09:11:38Z","timestamp":1627031498000},"page":"1245-1256","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["Maternal exposure to hydroxychloroquine and birth defects"],"prefix":"10.1002","volume":"113","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8420-5040","authenticated-orcid":false,"given":"Meredith M.","family":"Howley","sequence":"first","affiliation":[{"name":"Birth Defects Registry New York State Department of Health  Albany New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martha M","family":"Werler","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Boston University School of Public Health  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4702-1007","authenticated-orcid":false,"given":"Sarah C","family":"Fisher","sequence":"additional","affiliation":[{"name":"Birth Defects Registry New York State Department of Health  Albany New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alissa R","family":"Van Zutphen","sequence":"additional","affiliation":[{"name":"Birth Defects Registry New York State Department of Health  Albany New York USA"},{"name":"Department of Epidemiology and Biostatistics, School of Public Health University at Albany  Rensselaer New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Suzan L","family":"Carmichael","sequence":"additional","affiliation":[{"name":"Division of Neonatal and Developmental Medicine, Department of Pediatrics Stanford University School of Medicine  Stanford California USA"},{"name":"Division of Maternal\u2010Fetal Medicine and Obstetrics, Department of Obstetrics and Gynecology Stanford University School of Medicine  Stanford California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cheryl S","family":"Broussard","sequence":"additional","affiliation":[{"name":"Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Developmental Disabilities Centers for Disease Control and Prevention  Atlanta Georgia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9217-3723","authenticated-orcid":false,"given":"Dominique","family":"Heinke","sequence":"additional","affiliation":[{"name":"Center for Birth Defects Research and Prevention Massachusetts Department of Public Health  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth C","family":"Ailes","sequence":"additional","affiliation":[{"name":"Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Developmental Disabilities Centers for Disease Control and Prevention  Atlanta Georgia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shannon M","family":"Pruitt","sequence":"additional","affiliation":[{"name":"Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Developmental Disabilities Centers for Disease Control and Prevention  Atlanta Georgia USA"},{"name":"Oak Ridge Institute for Science and Education  Oak Ridge Tennessee USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennita","family":"Reefhuis","sequence":"additional","affiliation":[{"name":"Division of Birth Defects and Infant Disorders, National Center on Birth Defects and Developmental Disabilities Centers for Disease Control and Prevention  Atlanta Georgia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Allen A.","family":"Mitchell","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Boston University 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