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Electronic databases were searched and studies reporting pregnancy outcomes (termination, spontaneous abortion, demise, or live birth) for fetuses with 45,X karyotype and a CH diagnosed on ultrasound were included. For cases of survival, CH characteristics, presence of hydrops fetalis, or concomitant anomalies, delivery details, and postnatal outcomes were summarized. A total of 95 studies, including 535 cases, met inclusion criteria: 285 (53.3%) pregnancies were terminated, 72 (13.5%) had spontaneous abortion or demise, 164 (30.7%) had unspecified pregnancy failure, and 14 (2.6%) were live births. Among live births with data available, all CH measured 2\u20137 cm, more than half were septate, and almost all regressed in size or eventually disappeared. Hydrops fetalis was noted in five cases. Of the eight live births with neonatal outcomes available, three neonates died shortly after birth and five survived past the neonatal period. This review suggests that diagnosis of CH in a 45,X fetus is associated with an estimated live birth rate of 2.6%, but only 1% survive to infancy. Prognosis appears to improve with CH regression.<\/jats:p>","DOI":"10.1002\/ajmg.a.61902","type":"journal-article","created":{"date-parts":[[2020,10,7]],"date-time":"2020-10-07T16:45:31Z","timestamp":1602089131000},"page":"26-32","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Outcome of 45,X fetuses with cystic hygroma: A systematic review"],"prefix":"10.1002","volume":"185","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5348-9300","authenticated-orcid":false,"given":"Ariel T.","family":"Levy","sequence":"first","affiliation":[{"name":"Department of Obstetrics and Gynecology, Division of Maternal\u2010Fetal Medicine Thomas Jefferson University Hospital  Philadelphia Pennsylvania USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vincenzo","family":"Berghella","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Division of Maternal\u2010Fetal Medicine Thomas Jefferson University Hospital  Philadelphia Pennsylvania USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Huda B.","family":"Al\u2010Kouatly","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Division of Maternal\u2010Fetal Medicine Thomas Jefferson University Hospital  Philadelphia Pennsylvania USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2020,10,7]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/pd.1970090504"},{"issue":"4","key":"e_1_2_9_3_1","first-page":"487","article-title":"Hygroma kystique du cou: diagnostic antenatal, facteurs pronostiques, conduit \u00e0 tenir: A propos de 42 cas","volume":"20","author":"Bernard P.","year":"1991","journal-title":"Journal de Gyn\u00e9cologie Obst\u00e9trique et Biologie de la Reproduction"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/pd.1970110603"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/fn.74.1.F38"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/0002-9378(89)90237-8"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1067\/mob.2003.230"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-2007-994744"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0029-7844(96)00358-4"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0046-8177(84)80331-7"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajog.2014.12.018"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.3109\/00016349609033353"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198310063091403"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/pd.1970080408"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1097\/00006254-198906000-00025"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0301-2115(96)02590-0"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0095-5108(18)30560-8"},{"issue":"3","key":"e_1_2_9_18_1","first-page":"248","article-title":"Turner syndrome with hygroma colli and an unusually severe skeletal defect","volume":"25","author":"Ferrier P. 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