{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T17:21:03Z","timestamp":1790961663509,"version":"4.1.0"},"reference-count":29,"publisher":"Wiley","issue":"12","license":[{"start":{"date-parts":[[2022,6,29]],"date-time":"2022-06-29T00:00:00Z","timestamp":1656460800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2022,7,15]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Conjoined triplets are among the rarest of human malformations, as are asymmetric or parasitic conjoined twins. Based on a very modest corpus of recent literature, we applied the embryonic disk model of conjoined twinning to 10 previously reported cases involving asymmetric anatomical multiplications to determine whether they concerned conjoined twins or conjoined triplets. In spite of their phenotypic similarities, we diagnosed four of these cases as conjoined twins and three of them as conjoined triplets. In the remaining three cases, no definite diagnosis could be made, as essential information was lacking from the reports. We conclude that it is not necessarily the expected duplication or triplication of structures that points to the correct diagnosis in these cases, but the number and mutual position of the hearts they presented with. Considering their rarity we stress to thoroughly investigate and describe internal (dys)morphology in novel cases of (asymmetric) conjoined twins and triplets to further unravel their pathogenicity and come to the correct diagnoses.<\/jats:p>","DOI":"10.1002\/bdr2.2066","type":"journal-article","created":{"date-parts":[[2022,6,29]],"date-time":"2022-06-29T10:38:06Z","timestamp":1656499086000},"page":"596-610","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["Conjoined twins and conjoined triplets: At the heart of the matter"],"prefix":"10.1002","volume":"114","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2452-8307","authenticated-orcid":false,"given":"Roelof\u2010Jan","family":"Oostra","sequence":"first","affiliation":[{"name":"Department of Medical Biology, Section Clinical Anatomy &amp; Embryology, Amsterdam University Medical Centers, location Academic Medical Center University of Amsterdam  The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annelieke N.","family":"Schepens\u2010Franke","sequence":"additional","affiliation":[{"name":"Department of Imaging, Section Anatomy and Museum for Anatomy and Pathology Radboud University Medical Center  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giovanni","family":"Magno","sequence":"additional","affiliation":[{"name":"Department of Cardiac, Thoracic and Vascular Sciences, Section of Medical Humanities, Padua University Medical School and Museum of Pathological Anatomy University Museums Centre, University of Padua  Padua Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alberto","family":"Zanatta","sequence":"additional","affiliation":[{"name":"Department of Cardiac, Thoracic and Vascular Sciences, Section of Medical Humanities, Padua University Medical School and Museum of Pathological Anatomy University Museums Centre, University of Padua  Padua Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1564-0847","authenticated-orcid":false,"given":"Lucas L.","family":"Boer","sequence":"additional","affiliation":[{"name":"Department of Imaging, Section Anatomy and Museum for Anatomy and Pathology Radboud University Medical Center  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,6,29]]},"reference":[{"issue":"17","key":"e_1_2_6_2_1","first-page":"1451","article-title":"Compound monstrosity","volume":"154","author":"Anonymous","year":"1954","journal-title":"Journal of the American Medical Association"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajog.2004.10.622"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/ca.23387"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/ca.23725"},{"key":"e_1_2_6_6_1","volume-title":"Descrizione di una mostruosa bambina nata nel Veronese","author":"Bongiovanni Z.","year":"1789"},{"key":"e_1_2_6_7_1","first-page":"697","article-title":"Description d'un enfant monstreux n\u00e9 \u00e0 terme","author":"Bordenave M.","year":"1776","journal-title":"Mem de l'Acad des sciences"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/pd.1970030415"},{"key":"e_1_2_6_9_1","volume-title":"Monstri bicipitis descriptio anatomica: dissertatio inauguralis medica","author":"Hesse E.","year":"1823"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12098-010-0071-8"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.2350\/08-11-0555.1"},{"key":"e_1_2_6_12_1","unstructured":"Kaufmann E. Comalli Dillon K. Cuillier F. Lafitte A. &Ranjatoelina A.(2007).Abnormal triplet pregnancy. From the fetus net"},{"key":"e_1_2_6_13_1","first-page":"551","article-title":"Beschreibung einer Missgeburt","volume":"4","author":"Klein","year":"1818","journal-title":"Deut Arch f\u00fcr Phys"},{"key":"e_1_2_6_14_1","unstructured":"Magno G. Boer L. L. Oostra R.\u2010J. & Zanatta A. (2022).Conjoined twins in the anatomical collections of the University of Padua Italy. American Journal of Medical Genetics Part A. Manuscript submitted for publication.\u00a0"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30321"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1053\/jpsu.2000.16432"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/0002-9378(52)90220-2"},{"key":"e_1_2_6_18_1","volume-title":"Monstri duplicis per implantationem expositio anatomica","author":"Peter J. U.","year":"1844"},{"key":"e_1_2_6_19_1","volume-title":"Sopra un feto umano tricefalo: memoria ostetrica ad anatomic adel. letta all'Accademia Gioenia de Scienze Naturale nelle sedute ordinarie dei 16 febbraro et 15 marzo 1832 ed inserita nel vol. VIII degli atti accademici","author":"Reina E.","year":"1841"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2004.11.006"},{"issue":"9","key":"e_1_2_6_21_1","first-page":"931","article-title":"Four decades of conjoined twins at red cross Children's hospital\u2010\u2010lessons learned","volume":"96","author":"Rode H.","year":"2006","journal-title":"South African Medical Journal"},{"key":"e_1_2_6_22_1","volume-title":"Monstri duplicis rarissimi descriptio anatomica","author":"Rosenstiel A.","year":"1824"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2010.07.002"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/ca.1079"},{"key":"e_1_2_6_25_1","doi-asserted-by":"crossref","first-page":"1","DOI":"10.56021\/9780801870705","volume-title":"Conjoined twins: Developmental malformations and clinical implications","author":"Spencer R.","year":"2003"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420260203"},{"key":"e_1_2_6_27_1","volume-title":"Istoria della generazione dell'uomo, e degli animali, se sia da' vermicelli spermatici o dalle uova; con un trattato nel fine della sterilit\u00e0 e de' suoi rimedi","author":"Vallisneri A.","year":"1721"},{"key":"e_1_2_6_28_1","volume-title":"Duorum monstrorum duplicium humanorum descriptio anatomica","author":"Wirtensohn J.","year":"1825"},{"key":"e_1_2_6_29_1","volume-title":"Testo atlante di patologia nella storia. Dal Museo di Anatomia Patologi\u2010ca dell'Universit\u00e0 di Padova","author":"Zanatta A.","year":"2015"},{"key":"e_1_2_6_30_1","doi-asserted-by":"publisher","DOI":"10.1111\/cura.12273"}],"container-title":["Birth Defects Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/bdr2.2066","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/bdr2.2066","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/bdr2.2066","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,24]],"date-time":"2023-08-24T08:40:47Z","timestamp":1692866447000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/bdr2.2066"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,6,29]]},"references-count":29,"journal-issue":{"issue":"12","published-print":{"date-parts":[[2022,7,15]]}},"alternative-id":["10.1002\/bdr2.2066"],"URL":"https:\/\/doi.org\/10.1002\/bdr2.2066","archive":["Portico"],"relation":{},"ISSN":["2472-1727","2472-1727"],"issn-type":[{"value":"2472-1727","type":"print"},{"value":"2472-1727","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,6,29]]},"assertion":[{"value":"2022-02-24","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-06-01","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-06-29","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}