{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T08:47:58Z","timestamp":1777452478063,"version":"3.51.4"},"reference-count":33,"publisher":"Oxford University Press (OUP)","issue":"5","license":[{"start":{"date-parts":[[2025,3,10]],"date-time":"2025-03-10T00:00:00Z","timestamp":1741564800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,5,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Placental mesenchymal dysplasia (PMD) is a rare placental pathology that may be associated with Beckwith\u2013Wiedemann features in the fetus and may be due to the presence of an androgenetic cell line. Many of the reported PMD cases describe the presence of a biparental and an isodisomic androgenetic cell line. The proposed mechanism of formation is by fertilization of a haploid ovum by a haploid sperm and duplication of the male pronucleus. We present a case with evidence of the participation of three different haploid gametes, one ovum and two spermatozoa, which led to an androgenetic\/biparental chimera (ABC) in which three fetal cell lines were detected: two biparental, genetically different, cell lines but with the same maternal contribution, and one heterodisomic androgenetic cell line. To our knowledge, this is the first described case of ABC with two different biparental cell lines. We propose a novel mechanism based on the heterogoneic division of the tripronucleated zygote to explain the formation of this rare ABC.<\/jats:p>","DOI":"10.1093\/humrep\/deaf038","type":"journal-article","created":{"date-parts":[[2025,3,10]],"date-time":"2025-03-10T22:30:26Z","timestamp":1741645826000},"page":"962-970","source":"Crossref","is-referenced-by-count":3,"title":["Case Report: Androgenetic\/biparental chimera with two biparental cell lines leading to placental mesenchymal dysplasia: a possible novel mechanism of formation"],"prefix":"10.1093","volume":"40","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-1178-5310","authenticated-orcid":false,"given":"Mar","family":"Xuncl\u00e0","sequence":"first","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mar\u00eda \u00c1ngeles","family":"S\u00e1nchez-Dur\u00e1n","sequence":"additional","affiliation":[{"name":"Fetal Medicine Unit, Maternal-Fetal Medicine Department, Vall d\u2019Hebron University Hospital and Universitat Aut\u00f2noma de Barcelona , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nat\u00e0lia","family":"Rey","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mar\u00eda","family":"Serrano","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pedro Antonio","family":"Mart\u00ednez","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lourdes","family":"Trobo","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jessica","family":"Camacho Soriano","sequence":"additional","affiliation":[{"name":"Pathology Department, Vall d\u2019Hebron University Hospital , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alberto","family":"Plaja","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Neus","family":"Castells-Sarret","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria \u00c0ngels","family":"Rigola","sequence":"additional","affiliation":[{"name":"Department of Cellular Biology and Medical Genetics, Universitat Aut\u00f2noma de Barcelona , Bellaterra,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0096-3336","authenticated-orcid":false,"given":"Elena","family":"Garc\u00eda-Arum\u00ed","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eduardo Fidel","family":"Tizzano","sequence":"additional","affiliation":[{"name":"Department of Clinical and Molecular Genetics, Vall d\u2019Hebron University Hospital and Medicine Genetics Group, Vall d\u2019Hebron Research Institute , Barcelona,","place":["Spain"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2025,3,10]]},"reference":[{"key":"2025050201521754000_deaf038-B1","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1186\/s13148-022-01280-0","article-title":"Aberrant hypomethylation at imprinted differentially methylated regions is involved in biparental placental mesenchymal dysplasia","volume":"14","author":"Aoki","year":"2022","journal-title":"Clin Epigenetics"},{"key":"2025050201521754000_deaf038-B2","doi-asserted-by":"crossref","first-page":"309","DOI":"10.1111\/j.1741-4520.2002.tb00897.x","article-title":"Suspected involvement of the X chromosome in placental mesenchymal dysplasia","volume":"42","author":"Arizawa","year":"2002","journal-title":"Congenit Anom (Kyoto)"},{"key":"2025050201521754000_deaf038-B3","doi-asserted-by":"crossref","first-page":"519","DOI":"10.1097\/PAT.0b013e3283559c94","article-title":"The placenta in Beckwith-Wiedemann syndrome: genotype-phenotype associations, excessive extravillous trophoblast and placental mesenchymal dysplasia","volume":"44","author":"Armes","year":"2012","journal-title":"Pathology"},{"key":"2025050201521754000_deaf038-B4","doi-asserted-by":"crossref","first-page":"187","DOI":"10.1002\/pd.1103","article-title":"Placental mesenchymal dysplasia associated with fetal aneuploidy","volume":"25","author":"Cohen","year":"2005","journal-title":"Prenat Diagn"},{"key":"2025050201521754000_deaf038-B5","doi-asserted-by":"crossref","first-page":"6755","DOI":"10.1038\/s41467-022-34294-6","article-title":"The first mitotic division of human embryos is highly error prone","volume":"13","author":"Currie","year":"2022","journal-title":"Nat Commun"},{"key":"2025050201521754000_deaf038-B6","doi-asserted-by":"crossref","first-page":"201","DOI":"10.1186\/s13059-022-02763-2","article-title":"Parental genomes segregate into distinct blastomeres during multipolar zygotic divisions leading to mixoploid and chimeric blastocysts","volume":"23","author":"De Coster","year":"2022","journal-title":"Genome Biol"},{"key":"2025050201521754000_deaf038-B7","doi-asserted-by":"publisher","DOI":"10.1002\/bies.201600226","article-title":"How can zygotes segregate entire parental genomes into distinct blastomeres? The zygote metaphase revisited","volume":"39","author":"Destouni","year":"2017","journal-title":"Bioessays"},{"key":"2025050201521754000_deaf038-B8","doi-asserted-by":"crossref","first-page":"567","DOI":"10.1101\/gr.200527.115","article-title":"Zygotes segregate entire parental genomes in distinct blastomere lineages causing cleavage-stage chimerism and mixoploidy","volume":"26","author":"Destouni","year":"2016","journal-title":"Genome Res"},{"key":"2025050201521754000_deaf038-B9","doi-asserted-by":"crossref","first-page":"157","DOI":"10.1080\/14647270600636269","article-title":"Can \u2018abnormally\u2019 fertilized zygotes give rise to viable embryos?","volume":"9","author":"Feenan","year":"2006","journal-title":"Hum Fertil (Camb)"},{"key":"2025050201521754000_deaf038-B10","doi-asserted-by":"crossref","first-page":"842","DOI":"10.1056\/NEJMoa1701313","article-title":"Molecular support for heterogonesis resulting in sesquizygotic twinning","volume":"380","author":"Gabbett","year":"2019","journal-title":"N Engl J Med"},{"key":"2025050201521754000_deaf038-B11","doi-asserted-by":"crossref","first-page":"1557","DOI":"10.1002\/ccr3.1662","article-title":"Discordance for placental mesenchymal dysplasia in a monochorionic diamniotic twin pregnancy: a case report","volume":"6","author":"Gheysen","year":"2018","journal-title":"Clin Case Rep"},{"key":"2025050201521754000_deaf038-B12","doi-asserted-by":"crossref","first-page":"236","DOI":"10.1093\/humrep\/deg060","article-title":"Postzygotic diploidization of triploids as a source of unusual cases of mosaicism, chimerism and twinning","volume":"18","author":"Golubovsky","year":"2003","journal-title":"Hum Reprod"},{"key":"2025050201521754000_deaf038-B13","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1016\/j.ejogrb.2019.01.014","article-title":"Placental mesenchymal dysplasia: an underdiagnosed placental pathology with various clinical outcomes","volume":"234","author":"Guenot","year":"2019","journal-title":"Eur J Obstet Gynecol Reprod Biol"},{"key":"2025050201521754000_deaf038-B14","doi-asserted-by":"crossref","first-page":"454","DOI":"10.1016\/j.placenta.2008.01.001","article-title":"Placental mesenchymal dysplasia with Beckwith-Wiedemann syndrome fetus in the context of biparental and androgenic cell lines","volume":"29","author":"H'mida","year":"2008","journal-title":"Placenta"},{"key":"2025050201521754000_deaf038-B16","doi-asserted-by":"crossref","first-page":"187","DOI":"10.1136\/jmg.2005.033571","article-title":"Androgenetic\/biparental mosaicism causes placental mesenchymal dysplasia","volume":"43","author":"Kaiser-Rogers","year":"2006","journal-title":"J Med Genet"},{"key":"2025050201521754000_deaf038-B17","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1016\/j.acthis.2014.11.009","article-title":"Tripolar mitosis in human cells and embryos: occurrence, pathophysiology and medical implications","volume":"117","author":"Kalatova","year":"2015","journal-title":"Acta Histochem"},{"key":"2025050201521754000_deaf038-B18","doi-asserted-by":"crossref","first-page":"103971","DOI":"10.1016\/j.ejmg.2020.103971","article-title":"Natural human chimeras: a review","volume":"63","author":"Madan","year":"2020","journal-title":"Eur J Med Genet"},{"key":"2025050201521754000_deaf038-B19","doi-asserted-by":"crossref","first-page":"554","DOI":"10.1002\/pd.5895","article-title":"Genome-wide abnormalities in embryos: origins and clinical consequences","volume":"41","author":"Masset","year":"2021","journal-title":"Prenat Diagn"},{"key":"2025050201521754000_deaf038-B20","doi-asserted-by":"crossref","first-page":"1101","DOI":"10.1002\/ajmg.a.63112","article-title":"Mosaic genome-wide paternal uniparental disomy after discordant results from primary fetal samples and cultured cells","volume":"191","author":"Mastromoro","year":"2023","journal-title":"Am J Med Genet A"},{"key":"2025050201521754000_deaf038-B15","volume-title":"An International System for Human Cytogenomic Nomenclature","author":"McGowan-Jordan J, Hastings RJ, Moore S."},{"key":"2025050201521754000_deaf038-B21","doi-asserted-by":"crossref","first-page":"393.e5","DOI":"10.1016\/j.fertnstert.2009.03.090","article-title":"Reproductive consequences of genome-wide paternal uniparental disomy mosaicism: description of two cases with different mechanisms of origin and pregnancy outcomes","volume":"92","author":"Morales","year":"2009","journal-title":"Fertil Steril"},{"key":"2025050201521754000_deaf038-B22","doi-asserted-by":"crossref","first-page":"324","DOI":"10.1016\/S0344-0338(11)80791-0","article-title":"Placental vascular anomaly with diffuse mesenchymal stem villous hyperplasia. A new clinico-pathological entity?","volume":"187","author":"Moscoso","year":"1991","journal-title":"Pathol Res Pract"},{"key":"2025050201521754000_deaf038-B23","doi-asserted-by":"crossref","first-page":"131","DOI":"10.5858\/2007-131-131-PMD","article-title":"Placental mesenchymal dysplasia","volume":"131","author":"Parveen","year":"2007","journal-title":"Arch Pathol Lab Med"},{"key":"2025050201521754000_deaf038-B24","doi-asserted-by":"crossref","first-page":"89","DOI":"10.1093\/humrep\/7.suppl_1.89","article-title":"Fertilization abnormalities in human in-vitro fertilization","volume":"7 Suppl 1","author":"Plachot","year":"1992","journal-title":"Hum Reprod"},{"key":"2025050201521754000_deaf038-B25","first-page":"591956","article-title":"Pregnancy with concomitant chorioangioma and placental mesenchymal dysplasia: a rare placental abnormality","volume":"2013","author":"Qichang","year":"2013","journal-title":"Case Rep Obstet Gynecol"},{"key":"2025050201521754000_deaf038-B26","doi-asserted-by":"crossref","first-page":"564","DOI":"10.1111\/cge.13431","article-title":"Biparental\/androgenetic mosaicism in a male with features of overgrowth and placental mesenchymal dysplasia","volume":"94","author":"Repnikova","year":"2018","journal-title":"Clin Genet"},{"key":"2025050201521754000_deaf038-B27","doi-asserted-by":"crossref","first-page":"1114","DOI":"10.1093\/humrep\/del462","article-title":"Origin and outcome of pregnancies affected by androgenetic\/biparental chimerism","volume":"22","author":"Robinson","year":"2007","journal-title":"Hum Reprod"},{"key":"2025050201521754000_deaf038-B28","doi-asserted-by":"crossref","first-page":"1238","DOI":"10.1002\/pd.1879","article-title":"Utility of microsatellite analysis in evaluation of pregnancies with placental mesenchymal dysplasia","volume":"27","author":"Schuetzle","year":"2007","journal-title":"Prenat Diagn"},{"key":"2025050201521754000_deaf038-B29","doi-asserted-by":"crossref","first-page":"5563","DOI":"10.3390\/cancers14225563","article-title":"Placental mesenchymal dysplasia and Beckwith-Wiedemann syndrome","volume":"14","author":"Soejima","year":"2022","journal-title":"Cancers (Basel)"},{"key":"2025050201521754000_deaf038-B30","doi-asserted-by":"crossref","first-page":"1026","DOI":"10.1038\/ejhg.2011.93","article-title":"Mosaics and moles","volume":"19","author":"Sunde","year":"2011","journal-title":"Eur J Hum Genet"},{"key":"2025050201521754000_deaf038-B31","doi-asserted-by":"crossref","first-page":"1048","DOI":"10.1002\/pd.1255","article-title":"Twin pregnancy with a chimeric androgenetic and biparental placenta in one twin displaying placental mesenchymal dysplasia phenotype","volume":"25","author":"Surti","year":"2005","journal-title":"Prenat Diagn"},{"key":"2025050201521754000_deaf038-B32","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1038\/jhg.2010.142","article-title":"Androgenetic\/biparental mosaicism in a girl with Beckwith-Wiedemann syndrome-like and upd(14)pat-like phenotypes","volume":"56","author":"Yamazawa","year":"2011","journal-title":"J Hum Genet"},{"key":"2025050201521754000_deaf038-B33","doi-asserted-by":"crossref","first-page":"1562","DOI":"10.1002\/ajmg.a.62665","article-title":"Prenatal presentation in two fetuses with features of Beckwith Wiedemann syndrome\u2014an unexpected diagnosis of androgenetic chimera and its clinical implications","volume":"188","author":"Yu","year":"2022","journal-title":"Am J Med Genet A"}],"container-title":["Human Reproduction"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/humrep\/article-pdf\/40\/5\/962\/62369935\/deaf038.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/humrep\/article-pdf\/40\/5\/962\/62369935\/deaf038.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,5,2]],"date-time":"2025-05-02T05:52:49Z","timestamp":1746165169000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/humrep\/article\/40\/5\/962\/8068974"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3,10]]},"references-count":33,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2025,3,10]]},"published-print":{"date-parts":[[2025,5,1]]}},"URL":"https:\/\/doi.org\/10.1093\/humrep\/deaf038","relation":{},"ISSN":["0268-1161","1460-2350"],"issn-type":[{"value":"0268-1161","type":"print"},{"value":"1460-2350","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2025,5]]},"published":{"date-parts":[[2025,3,10]]}}}