{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,27]],"date-time":"2026-10-27T21:54:07Z","timestamp":1793138047951,"version":"4.5.0"},"reference-count":25,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2022,3,3]],"date-time":"2022-03-03T00:00:00Z","timestamp":1646265600000},"content-version":"vor","delay-in-days":2,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["obgyn.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Ultrasound in Obstet &amp;amp; Gyne"],"published-print":{"date-parts":[[2022,3]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>To assess the rate and type of genetic diagnosis and clinical outcome of cases of fetal cardiomyopathy (CM) during two eras, in order to examine the impact of advanced genetic testing and improved perinatal management strategies.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>All diagnoses of fetal CM in Alberta, Canada, encountered between 2003 and 2019, were reviewed retrospectively. Genetic, cardiac and non\u2010cardiac diagnoses and clinical outcome were documented. Cases with CM secondary to extracardiac pathology and maternal conditions were excluded. Cases diagnosed in the earlier era of the study period (2003\u20132012) were compared with those diagnosed in the later era (2013\u20132019).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Thirty\u2010eight cases of fetal CM met the inclusion criteria. Median gestational age (GA) at diagnosis was 22.8 (range, 13.4\u201337.4) weeks. Associated structural heart disease was present in 39% (15\/38) of cases and 24% (9\/38) had arrhythmia. Hydrops was identified in 29% (11\/38) of cases at presentation, and a further 18% (7\/38) developed hydrops later in gestation. Twenty\u2010six percent (10\/38) of cases underwent termination of pregnancy and 24% (9\/38) had intrauterine death. Of liveborn cases,  neonatal death occurred in 16% (3\/19), late death occurred in 21% (4\/19) and 63% (12\/19) were alive at the last follow\u2010up. Excluding cases that had termination of pregnancy and those with a liveborn infant who received planned palliative care, the rate of neonatal survival was higher in the later compared with the earlier era (69% (11\/16) <jats:italic>vs<\/jats:italic> 45% (5\/11)), although the difference was not statistically significant (<jats:italic>P<\/jats:italic>\u2009=\u20090.26). A genetic etiology was confirmed in 39% (15\/38) of cases and strongly suspected in 24% (9\/38). A significantly higher proportion of cases had a confirmed or strongly suspected genetic etiology in the later era compared with in the earlier era (76% (19\/25) <jats:italic>vs<\/jats:italic> 38% (5\/13); <jats:italic>P<\/jats:italic>\u2009=\u20090.04).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>In the recent era, a higher proportion of fetal CM cases had a confirmed or strongly suspected genetic etiology than reported previously. Based on comparison with older series, modern perinatal management strategies may not have a significant impact on neonatal survival in cases of fetal CM; however, a larger study would be better powered to detect more subtle differences. \u00a9 2021 International Society of Ultrasound in Obstetrics and Gynecology.<\/jats:p><\/jats:sec>","DOI":"10.1002\/uog.23713","type":"journal-article","created":{"date-parts":[[2021,6,23]],"date-time":"2021-06-23T06:50:07Z","timestamp":1624431007000},"page":"325-334","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Clinical presentation, genetic etiology and outcome associated with fetal cardiomyopathy: comparison of two eras"],"prefix":"10.1002","volume":"59","author":[{"given":"T.","family":"Trakmulkichkarn","sequence":"first","affiliation":[{"name":"Fetal and Neonatal Cardiology and Heart Function and Transplant Programs, Division of Cardiology, Department of Pediatrics University of Alberta  Edmonton AB Canada"},{"name":"Women's and Children's Health Research Institute University of Alberta  Edmonton AB Canada"},{"name":"Department of Obstetrics and Gynecology Phramongkutklao Hospital  Bangkok Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R.","family":"Ghadiry\u2010Tavi","sequence":"additional","affiliation":[{"name":"Cumming School of Medicine, University of Calgary, Department of Pediatrics Alberta Children's Hospital  Calgary AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D.","family":"Fruitman","sequence":"additional","affiliation":[{"name":"Cumming School of Medicine, University of Calgary, Department of Pediatrics Alberta Children's Hospital  Calgary AB Canada"},{"name":"Section of Cardiology Alberta Children's Hospital  Calgary AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K. Y.","family":"Niederhoffer","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics University of Alberta  Edmonton AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"O.","family":"Caluseriu","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics University of Alberta  Edmonton AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. L.","family":"Lauzon","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics Alberta Children's Hospital  Calgary AB Canada"},{"name":"Alberta Children's Hospital Research Institute for Child and Maternal Health Alberta Children's Hospital  Calgary AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G.","family":"Wewala","sequence":"additional","affiliation":[{"name":"Fetal and Neonatal Cardiology and Heart Function and Transplant Programs, Division of Cardiology, Department of Pediatrics University of Alberta  Edmonton AB Canada"},{"name":"Women's and Children's Health Research Institute University of Alberta  Edmonton AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L. K.","family":"Hornberger","sequence":"additional","affiliation":[{"name":"Fetal and Neonatal Cardiology and Heart Function and Transplant Programs, Division of Cardiology, Department of Pediatrics University of Alberta  Edmonton AB Canada"},{"name":"Women's and Children's Health Research Institute University of Alberta  Edmonton AB Canada"},{"name":"Department of Obstetrics and Gynecology University of Alberta  Edmonton AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Urschel","sequence":"additional","affiliation":[{"name":"Fetal and Neonatal Cardiology and Heart Function and Transplant Programs, Division of Cardiology, Department of Pediatrics University of Alberta  Edmonton AB Canada"},{"name":"Women's and Children's Health Research Institute University of Alberta  Edmonton AB Canada"},{"name":"Department of Medical Microbiology and Immunology University of Alberta  Edmonton AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J.","family":"Conway","sequence":"additional","affiliation":[{"name":"Fetal and Neonatal Cardiology and Heart Function and Transplant Programs, Division of Cardiology, Department of Pediatrics University of Alberta  Edmonton AB Canada"},{"name":"Women's and Children's Health Research Institute University of Alberta  Edmonton AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9733-5795","authenticated-orcid":false,"given":"A.","family":"McBrien","sequence":"additional","affiliation":[{"name":"Fetal and Neonatal Cardiology and Heart Function and Transplant Programs, Division of Cardiology, Department of Pediatrics University of Alberta  Edmonton AB Canada"},{"name":"Women's and Children's Health Research Institute University of Alberta  Edmonton AB Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,3,3]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa021715"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1017\/S1047951105000855"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1161\/01.CIR.0000023900.58293.FE"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1155\/2010\/628451"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1161\/01.cir.0000437597.44550.5d"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.pcsu.2017.11.005"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.healun.2014.09.018"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/MOP.0000000000000398"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.7863\/ultra.15.05059"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jchf.2014.02.010"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.echo.2006.05.027"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/uog.6309"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/uog.7510"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/pd.1612"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/uog.12279"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00246-015-1250-1"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijcard.2020.12.013"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00246-015-1200-y"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00246-016-1369-8"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1136\/hrt.2009.165514"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.amjcard.2005.05.063"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.amjcard.2018.04.040"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ahj.2012.04.018"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1080\/14767058.2017.1342804"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.echo.2011.09.019"}],"container-title":["Ultrasound in Obstetrics &amp; 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