{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T14:50:52Z","timestamp":1786978252102,"version":"build-2736575974"},"reference-count":27,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2024,7,23]],"date-time":"2024-07-23T00:00:00Z","timestamp":1721692800000},"content-version":"vor","delay-in-days":22,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Molec Gen &amp; Gen Med"],"published-print":{"date-parts":[[2024,7]]},"abstract":"Abstract<\/jats:title>\n \n Background<\/jats:title>\n \n Glucose\u20106\u2010phosphate dehydrogenase (\n G6PD<\/jats:italic>\n ) deficiency is an X\u2010linked recessive disorder that predisposes individuals to hemolysis due to an inborn error of metabolism. We performed a systematic literature review to evaluate\n G6PD<\/jats:italic>\n deficiency as a possible etiology of nonimmune hydrops fetalis (NIHF) and severe fetal anemia.\n <\/jats:p>\n <\/jats:sec>\n \n Methods<\/jats:title>\n \n PubMed, OVID Medline, Scopus, and\n clinicaltrials.gov<\/jats:ext-link>\n were queried from inception until 31 April 2023 for all published cases of NIHF and severe fetal anemia caused by\n G6PD<\/jats:italic>\n deficiency. Keywords included \u201cfetal edema,\u201d \u201chydrops fetalis,\u201d \u201cglucose 6 phosphate dehydrogenase deficiency,\u201d and \u201cfetal anemia.\u201d Cases with workup presuming G6PD deficiency as an etiology for NIHF and severe fetal anemia were included. PRISMA guidelines were followed.\n <\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n Five cases of\n G6PD<\/jats:italic>\n \u2010related NIHF and one case of severe fetal anemia were identified. Four fetuses (4\/6, 66.7%) were male and two fetuses (2\/6, 33.3%) were female. Mean gestational age at diagnosis of NIHF\/anemia and delivery was 32.2\u2009\u00b1\u20094.9 and 35.7\u2009\u00b1\u20092.4\u2009weeks, respectively. Four cases (66.7%) required a cordocentesis for fetal transfusion, and two cases (33.3%) received blood transfusions immediately following delivery. Among the four multigravida cases, two (50%) noted previous pregnancies complicated by neonatal anemia. When reported, the maternal cases included two\n G6PD<\/jats:italic>\n deficiency carrier patients and two\n G6PD<\/jats:italic>\n \u2010deficient patients. Exposures to substances known to cause\n G6PD<\/jats:italic>\n deficiency\u2010related hemolysis occurred in 3\/6 (50%) cases.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusion<\/jats:title>\n \n Six cases of NIHF\/severe fetal anemia were associated with\n G6PD<\/jats:italic>\n deficiency. While\n G6PD<\/jats:italic>\n deficiency is an X\u2010linked recessive condition, female fetuses can be affected. Fetal\n G6PD<\/jats:italic>\n deficiency testing can be considered if parental history indicates, particularly if the standard workup for NIHF is negative.\n <\/jats:p>\n <\/jats:sec>","DOI":"10.1002\/mgg3.2491","type":"journal-article","created":{"date-parts":[[2024,7,23]],"date-time":"2024-07-23T11:43:43Z","timestamp":1721735023000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Glucose\u20106\u2010phosphate dehydrogenase deficiency as a cause for nonimmune hydrops fetalis and severe fetal anemia: A systematic review"],"prefix":"10.1002","volume":"12","author":[{"given":"Neel S.","family":"Iyer","sequence":"first","affiliation":[{"name":"Division of Maternal\u2010Fetal Medicine, Department of Obstetrics and Gynecology, Sidney Kimmel Medical College Thomas Jefferson University Philadelphia Pennsylvania USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1717-5934","authenticated-orcid":false,"given":"Matthew H.","family":"Mossayebi","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology Brigham and Women's Hospital, Harvard Medical School Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9342-7326","authenticated-orcid":false,"given":"Tracy J.","family":"Gao","sequence":"additional","affiliation":[{"name":"Sidney Kimmel Medical College Thomas Jefferson University Philadelphia Pennsylvania USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lylach","family":"Haizler\u2010Cohen","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology MedStar Washington Hospital Center Washington DC USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniele","family":"Di\u00a0Mascio","sequence":"additional","affiliation":[{"name":"Department of Maternal and Child Health and Urological Sciences Sapienza University of Rome Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5013-3456","authenticated-orcid":false,"suffix":"Jr","given":"Rodney A.","family":"McLaren","sequence":"additional","affiliation":[{"name":"Division of Maternal\u2010Fetal Medicine, Department of Obstetrics and Gynecology, Sidney Kimmel Medical College Thomas Jefferson University Philadelphia Pennsylvania USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2922-0333","authenticated-orcid":false,"given":"Huda B.","family":"Al\u2010Kouatly","sequence":"additional","affiliation":[{"name":"Division of Maternal\u2010Fetal Medicine, Department of Obstetrics and Gynecology, Sidney Kimmel Medical College Thomas Jefferson University Philadelphia Pennsylvania USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,7,23]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2012.02.045"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.114.1.297"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(08)60073-2"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1201\/9781003099062-56"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tips.2021.07.002"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2023.01.003"},{"key":"e_1_2_11_8_1","unstructured":"Genetics Home Reference U.S. National Library of Medicine. 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