{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T00:21:26Z","timestamp":1790814086482,"version":"4.1.0"},"reference-count":53,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2018,5,15]],"date-time":"2018-05-15T00:00:00Z","timestamp":1526342400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["American J of Med Genetics Pt C"],"published-print":{"date-parts":[[2018,6]]},"abstract":"<jats:p>Holoprosencephaly (HPE) is a major structural birth defect of the brain that occurs in approximately 1 in 10,000 live births. Although some genetic causes of HPE are known, a substantial proportion of cases have an unknown etiology. Due to the low birth prevalence and rarity of exposure to many potential risk factors for HPE, few epidemiologic studies have had sufficient sample size to examine risk factors. A 2010 review of the literature identified several risk factors that had been consistently identified as occurring more frequently among cases of HPE, including maternal diabetes, twinning, and a predominance of females, while also identifying a number of potential risk factors that had been less widely studied. In this article, we summarize a systematic literature review conducted to update the evidence for nongenetic risk factors for HPE.<\/jats:p>","DOI":"10.1002\/ajmg.c.31614","type":"journal-article","created":{"date-parts":[[2018,5,15]],"date-time":"2018-05-15T07:37:38Z","timestamp":1526369858000},"page":"151-164","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":28,"title":["Nongenetic risk factors for holoprosencephaly: An updated review of the epidemiologic literature"],"prefix":"10.1002","volume":"178","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1195-1261","authenticated-orcid":false,"given":"April D.","family":"Summers","sequence":"first","affiliation":[{"name":"Division of Congenital and Developmental Disorders National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention Atlanta Georgia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennita","family":"Reefhuis","sequence":"additional","affiliation":[{"name":"Division of Congenital and Developmental Disorders National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention Atlanta Georgia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joanna","family":"Taliano","sequence":"additional","affiliation":[{"name":"LAC Group, Contractor to US Centers for Disease Control and Prevention, Library Science Branch, Division of Public Health Information Dissemination Center for Surveillance, Epidemiology, and Laboratory Services Atlanta Georgia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sonja A.","family":"Rasmussen","sequence":"additional","affiliation":[{"name":"Office of Infectious Diseases Centers for Disease Control and Prevention Atlanta Georgia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2018,5,15]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.ede.0000147122.97061.bb"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1159\/000263552"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.018275"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20573"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1067\/mob.2001.111071"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20749"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tjog.2012.09.021"},{"issue":"5","key":"e_1_2_7_9_1","doi-asserted-by":"crossref","first-page":"1146","DOI":"10.1542\/peds.111.S1.1146","article-title":"Do multivitamin supplements attenuate the risk for diabetes\u2010associated birth defects?","volume":"111","author":"Correa A.","year":"2003","journal-title":"Pediatrics"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajog.2008.06.028"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajog.2011.12.018"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1001\/archpediatrics.2009.188"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(20000214)90:4<320::AID-AJMG11>3.0.CO;2-8"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1136\/jech-2015-206302"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.4239\/wjd.v6.i3.481"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ntt.2015.12.004"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23049"},{"issue":"5","key":"e_1_2_7_18_1","first-page":"AD01","article-title":"Agnathia holoprosencephaly and situs inversus in a neonate born to an alcoholic mother","volume":"9","author":"Goswami D.","year":"2015","journal-title":"Journal of Clinical and Diagnostic Research"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30238"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0176440"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23527"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.1095"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajog.2016.07.022"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30242"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.29.1.30"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19980630)78:2<140::AID-AJMG8>3.0.CO;2-S"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420160304"},{"key":"e_1_2_7_28_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36865"},{"key":"e_1_2_7_29_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30244"},{"key":"e_1_2_7_30_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19980501)77:2<139::AID-AJMG6>3.0.CO;2-N"},{"key":"e_1_2_7_31_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19971212)73:2<217::AID-AJMG20>3.0.CO;2-S"},{"key":"e_1_2_7_32_1","first-page":"301","article-title":"Pathogenetic analysis of congenital anomalies in humans","volume":"12","author":"Opitz J. M.","year":"1982","journal-title":"Pathobiology Annual"},{"key":"e_1_2_7_33_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30323"},{"key":"e_1_2_7_34_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.32104"},{"key":"e_1_2_7_35_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30233"},{"key":"e_1_2_7_36_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdrc.21090"},{"key":"e_1_2_7_37_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13256-016-1141-y"},{"key":"e_1_2_7_38_1","doi-asserted-by":"publisher","DOI":"10.1093\/aje\/kws201"},{"key":"e_1_2_7_39_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.32077"},{"key":"e_1_2_7_40_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19961218)66:3<320::AID-AJMG16>3.0.CO;2-O"},{"key":"e_1_2_7_41_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.10012"},{"key":"e_1_2_7_42_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23384"},{"key":"e_1_2_7_43_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30236"},{"key":"e_1_2_7_44_1","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2014.00040"},{"key":"e_1_2_7_45_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.10150"},{"key":"e_1_2_7_46_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23218"},{"key":"e_1_2_7_47_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23058"},{"key":"e_1_2_7_48_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320280114"},{"key":"e_1_2_7_49_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.22846"},{"key":"e_1_2_7_50_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-3016.2011.01215.x"},{"key":"e_1_2_7_51_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.35240"},{"key":"e_1_2_7_52_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.siny.2010.06.002"},{"key":"e_1_2_7_53_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.33.7.578"},{"key":"e_1_2_7_54_1","doi-asserted-by":"publisher","DOI":"10.1093\/phr\/116.S1.32"}],"container-title":["American Journal of Medical Genetics Part C: Seminars in Medical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.c.31614","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.c.31614","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/ajmg.c.31614","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.c.31614","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,10]],"date-time":"2023-09-10T15:14:37Z","timestamp":1694358877000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.c.31614"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,5,15]]},"references-count":53,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2018,6]]}},"alternative-id":["10.1002\/ajmg.c.31614"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.c.31614","archive":["Portico"],"relation":{},"ISSN":["1552-4868","1552-4876"],"issn-type":[{"value":"1552-4868","type":"print"},{"value":"1552-4876","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,5,15]]},"assertion":[{"value":"2018-03-16","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-04-04","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-05-15","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}