{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T08:48:19Z","timestamp":1777452499086,"version":"3.51.4"},"reference-count":88,"publisher":"MDPI AG","issue":"22","license":[{"start":{"date-parts":[[2022,11,12]],"date-time":"2022-11-12T00:00:00Z","timestamp":1668211200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Japan Society for the Promotion of Science (KAKENHI)","award":["JP21K19451"],"award-info":[{"award-number":["JP21K19451"]}]},{"name":"Japan Society for the Promotion of Science (KAKENHI)","award":["JP20H03643"],"award-info":[{"award-number":["JP20H03643"]}]},{"name":"Japan Society for the Promotion of Science (KAKENHI)","award":["JP20K08183"],"award-info":[{"award-number":["JP20K08183"]}]},{"name":"Japan Society for the Promotion of Science (KAKENHI)","award":["JP20ek0109486"],"award-info":[{"award-number":["JP20ek0109486"]}]},{"name":"Japan Society for the Promotion of Science (KAKENHI)","award":["JP20ek0109489"],"award-info":[{"award-number":["JP20ek0109489"]}]},{"name":"Japan Society for the Promotion of Science (KAKENHI)","award":["JP20FC1046"],"award-info":[{"award-number":["JP20FC1046"]}]},{"name":"Japan Agency for Medical Research and Development","award":["JP21K19451"],"award-info":[{"award-number":["JP21K19451"]}]},{"name":"Japan Agency for Medical Research and Development","award":["JP20H03643"],"award-info":[{"award-number":["JP20H03643"]}]},{"name":"Japan Agency for Medical Research and Development","award":["JP20K08183"],"award-info":[{"award-number":["JP20K08183"]}]},{"name":"Japan Agency for Medical Research and Development","award":["JP20ek0109486"],"award-info":[{"award-number":["JP20ek0109486"]}]},{"name":"Japan Agency for Medical Research and Development","award":["JP20ek0109489"],"award-info":[{"award-number":["JP20ek0109489"]}]},{"name":"Japan Agency for Medical Research and Development","award":["JP20FC1046"],"award-info":[{"award-number":["JP20FC1046"]}]},{"name":"Ministry of Health, Labor, and Welfare Program","award":["JP21K19451"],"award-info":[{"award-number":["JP21K19451"]}]},{"name":"Ministry of Health, Labor, and Welfare Program","award":["JP20H03643"],"award-info":[{"award-number":["JP20H03643"]}]},{"name":"Ministry of Health, Labor, and Welfare Program","award":["JP20K08183"],"award-info":[{"award-number":["JP20K08183"]}]},{"name":"Ministry of Health, Labor, and Welfare Program","award":["JP20ek0109486"],"award-info":[{"award-number":["JP20ek0109486"]}]},{"name":"Ministry of Health, Labor, and Welfare Program","award":["JP20ek0109489"],"award-info":[{"award-number":["JP20ek0109489"]}]},{"name":"Ministry of Health, Labor, and Welfare Program","award":["JP20FC1046"],"award-info":[{"award-number":["JP20FC1046"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cancers"],"abstract":"<jats:p>Placental mesenchymal dysplasia (PMD) is characterized by placentomegaly, aneurysmally dilated chorionic plate vessels, thrombosis of the dilated vessels, and large grapelike vesicles, and is often mistaken for partial or complete hydatidiform mole with a coexisting normal fetus. Androgenetic\/biparental mosaicism (ABM) has been found in many PMD cases. Beckwith\u2013Wiedemann syndrome (BWS) is an imprinting disorder with complex and diverse phenotypes and an increased risk of developing embryonal tumors. There are five major causative alterations: loss of methylation of imprinting control region 2 (KCNQ1OT1:TSS-DMR) (ICR2-LOM), gain of methylation at ICR1 (H19\/IGF2:IG-DMR) (ICR1-GOM), paternal uniparental disomy of 11 (pUPD11), loss-of-function variants of the CDKN1C gene, and paternal duplication of 11p15. Additional minor alterations include genetic variants within ICR1, paternal uniparental diploidy\/biparental diploidy mosaicism (PUDM, also called ABM), and genetic variants of KCNQ1. ABM (PUDM) is found in both conditions, and approximately 20% of fetuses from PMD cases are BWS and vice versa, suggesting a molecular link. PMD and BWS share some molecular characteristics in some cases, but not in others. These findings raise questions concerning the timing of the occurrence of the molecularly abnormal cells during the postfertilization period and the effects of these abnormalities on cell fates after implantation.<\/jats:p>","DOI":"10.3390\/cancers14225563","type":"journal-article","created":{"date-parts":[[2022,11,14]],"date-time":"2022-11-14T04:21:45Z","timestamp":1668399705000},"page":"5563","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Placental Mesenchymal Dysplasia and Beckwith\u2013Wiedemann Syndrome"],"prefix":"10.3390","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5076-6563","authenticated-orcid":false,"given":"Hidenobu","family":"Soejima","sequence":"first","affiliation":[{"name":"Division of Molecular Genetics and Epigenetics, Department of Biomolecular Sciences, Faculty of Medicine, Saga University, Saga 849-8501, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Satoshi","family":"Hara","sequence":"additional","affiliation":[{"name":"Division of Molecular Genetics and Epigenetics, Department of Biomolecular Sciences, Faculty of Medicine, Saga University, Saga 849-8501, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takashi","family":"Ohba","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Faculty of Life Sciences, Kumamoto University, Kumamoto 860-8556, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ken","family":"Higashimoto","sequence":"additional","affiliation":[{"name":"Division of Molecular Genetics and Epigenetics, Department of Biomolecular Sciences, Faculty of Medicine, Saga University, Saga 849-8501, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2022,11,12]]},"reference":[{"key":"ref_1","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. 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