{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T12:19:44Z","timestamp":1780489184944,"version":"3.54.1"},"reference-count":46,"publisher":"MDPI AG","issue":"10","license":[{"start":{"date-parts":[[2022,10,12]],"date-time":"2022-10-12T00:00:00Z","timestamp":1665532800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Children"],"abstract":"<jats:p>Conjoined twins represent a rare type of monoamniotic twins. Ultrasound assessment during the first trimester can facilitate the diagnosis, however further assessment by colour Doppler studies, 3D imaging, fetal echocardiography and fetal magnetic resonance imaging (MRI) is usually required in order to determine the specific fetal abnormalities and to guide appropriate pregnancy management. This case report presents a rare case of conjoined twins complicating a dichorionic-diamniotic triplet pregnancy, achieved after intracytoplasmic sperm injection (ICSI) and blastocyst transfer. A 44-year-old woman was referred for chorionicity determination to our Fetal Medicine Centre due to suspicion of conjoined twins in a triplet pregnancy. Ultrasound assessment at 11 weeks demonstrated a dichorionic triplet pregnancy which was also complicated by a rare type of conjoined twins (thoracoomphalopagus) and after a successful embryo reduction a neonate of 2200 g was delivered by caesarean section at term. The accurate diagnosis and early detection of conjoined twins by a fetal medicine specialist is crucial, especially as far as multiple pregnancies with three or more fetuses are concerned.<\/jats:p>","DOI":"10.3390\/children9101549","type":"journal-article","created":{"date-parts":[[2022,10,12]],"date-time":"2022-10-12T20:53:29Z","timestamp":1665608009000},"page":"1549","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Conjoined Twins Complicating a Dichorionic Triplet Pregnancy after Intracytoplasmic Sperm Injection: A Case Report and Review of the Literature"],"prefix":"10.3390","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0299-1317","authenticated-orcid":false,"given":"Anna","family":"Eleftheriades","sequence":"first","affiliation":[{"name":"Postgraduate Programme \u201cFetal Maternal Medicine\u2019\u2019, Medical School, National and Kapodistrian University of Athens, 11527 Athens, Greece"},{"name":"Department of Obstetrics and Gynaecology, Hospital of Langenthal\u2014Region Oberaargau, 4900 Langenthal, Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4112-8396","authenticated-orcid":false,"given":"Panagiotis","family":"Christopoulos","sequence":"additional","affiliation":[{"name":"Second Department of Obstetrics and Gynaecology, \u201cAretaieion\u2019\u2019 University Hospital, Medical School, National and Kapodistrian University of Athens, 11528 Athens, Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elsa","family":"Tsapakis","sequence":"additional","affiliation":[{"name":"Second Department of Obstetrics and Gynaecology, \u201cAretaieion\u2019\u2019 University Hospital, Medical School, National and Kapodistrian University of Athens, 11528 Athens, Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4854-734X","authenticated-orcid":false,"given":"Ermioni","family":"Tsarna","sequence":"additional","affiliation":[{"name":"Second Department of Obstetrics and Gynaecology, \u201cAretaieion\u2019\u2019 University Hospital, Medical School, National and Kapodistrian University of Athens, 11528 Athens, Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8479-6517","authenticated-orcid":false,"given":"Nikolaos F.","family":"Vlahos","sequence":"additional","affiliation":[{"name":"Second Department of Obstetrics and Gynaecology, \u201cAretaieion\u2019\u2019 University Hospital, Medical School, National and Kapodistrian University of Athens, 11528 Athens, Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1444-6161","authenticated-orcid":false,"given":"Emmanouil","family":"Kalampokas","sequence":"additional","affiliation":[{"name":"Second Department of Obstetrics and Gynaecology, \u201cAretaieion\u2019\u2019 University Hospital, Medical School, National and Kapodistrian University of Athens, 11528 Athens, Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6780-2687","authenticated-orcid":false,"given":"Daniele","family":"Bolla","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynaecology, Hospital of Langenthal\u2014Region Oberaargau, 4900 Langenthal, Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4967-2505","authenticated-orcid":false,"given":"Makarios","family":"Eleftheriades","sequence":"additional","affiliation":[{"name":"Second Department of Obstetrics and Gynaecology, \u201cAretaieion\u2019\u2019 University Hospital, Medical School, National and Kapodistrian University of Athens, 11528 Athens, Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2022,10,12]]},"reference":[{"key":"ref_1","first-page":"8","article-title":"Births: Final Data for 2020 Figure 1. Live Births and General Fertility Rates: United States","volume":"70","author":"Osterman","year":"2022","journal-title":"Natl. Vital Stat. Rep."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"274","DOI":"10.1002\/ajmg.c.30321","article-title":"Conjoined Twins: A Worldwide Collaborative Epidemiological Study of the International Clearinghouse for Birth Defects Surveillance and Research","volume":"157","author":"Mutchinick","year":"2011","journal-title":"Am. J. Med. Genet. Part C Semin. Med. Genet."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"814","DOI":"10.4103\/0377-4929.72100","article-title":"Dicephalus Dipus Tribrachius: A Case Report of Unusual Conjoined Twins","volume":"53","author":"Aparna","year":"2010","journal-title":"Indian J. Pathol. Microbiol."},{"key":"ref_4","first-page":"18","article-title":"The Ultrasonographic Prenatal Diagnosis of Conjoined Twins. A Case Report","volume":"58","author":"Azar","year":"2010","journal-title":"J. Med. Liban"},{"key":"ref_5","unstructured":"Nyberg, D., McGahan, J., Pretorius, D., and Pilu, G. (2003). Multiple Gestations. Diagnostic Imaging of Fetal Anomalies, Lippincott Williams & Wilkins."},{"key":"ref_6","unstructured":"Cunningham, F.G., Leveno, K.J., Bloom, S.L., Dashe, J.S., Hoffman, B.L., Casey, B.M., and Spong, C.Y. (2018). Multifetal Pregnancy. Williams Obstetrics, McGraw Hill Medical."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1159\/000132407","article-title":"Conjoined Twins in a Trichorionic Quadruplet Pregnancy after Ovulation Induction with Clomiphene Citrate","volume":"24","author":"Mendilcioglu","year":"2008","journal-title":"Fetal Diagn. Ther."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1002\/uog.185","article-title":"Conjoined Twins in a Triplet Pregnancy: Early Prenatal Diagnosis with Three-Dimensional Ultrasound and Review of the Literature","volume":"22","author":"Sepulveda","year":"2003","journal-title":"Ultrasound Obstet. Gynecol."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"820","DOI":"10.1002\/pd.1267","article-title":"First-Trimester Diagnosis of Conjoined Twins","volume":"25","author":"Pajkrt","year":"2005","journal-title":"Prenat. Diagn."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"387","DOI":"10.1007\/s00404-010-1440-2","article-title":"Cephalothoracopagus, Janiceps, Disymmetros, Monoomphalian Conjoined Twins Undiagnosed until Early Second Trimester","volume":"283","author":"Tonni","year":"2011","journal-title":"Arch. Gynecol. Obstet."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"249","DOI":"10.1097\/RUQ.0b013e31818c8858","article-title":"The Imaging Spectrum of Conjoined Twins","volume":"24","author":"Winkler","year":"2008","journal-title":"Ultrasound Q."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"252","DOI":"10.1007\/s00246-011-0125-3","article-title":"Conjoined Hearts in Thoracopagus Twins","volume":"33","author":"Collins","year":"2011","journal-title":"Pediatr. Cardiol."},{"key":"ref_13","first-page":"455","article-title":"Conoined Twins in a Triplet Pregnancy","volume":"122","author":"Tan","year":"1971","journal-title":"Am. J. Dis. Child."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"1413","DOI":"10.1093\/humrep\/15.6.1413","article-title":"First Trimester Diagnosis of Conjoined Twins in a Triplet Pregnancy after IVF and ICSI: Case Report","volume":"15","author":"Goldberg","year":"2000","journal-title":"Hum. Reprod."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"893","DOI":"10.1016\/S0002-9378(99)70321-2","article-title":"Selective Termination for Structural, Chromosomal, and Mendelian Anomalies: International Experience","volume":"181","author":"Evans","year":"1999","journal-title":"Am. J. Obstet. Gynecol."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"691","DOI":"10.1111\/j.1540-8175.2008.00854.x","article-title":"Echocardiographic Assessment of Conjoined Twins with Congenital Heart Disease in Shanghai","volume":"26","author":"Zhang","year":"2009","journal-title":"Echocardiography"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"941","DOI":"10.1016\/S0022-3468(96)90417-0","article-title":"Anatomic Description of Conjoined Twins: A Plea for Standardized Terminology","volume":"31","author":"Spencer","year":"1996","journal-title":"J. Pediatr. Surg."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"1043","DOI":"10.1056\/NEJM198804213181607","article-title":"Selective Reduction of Multifetal Pregnancies in the First Trimester","volume":"318","author":"Berkowitz","year":"1988","journal-title":"N. Engl. J. Med."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"90","DOI":"10.1016\/0140-6736(90)90550-O","article-title":"Selective Reduction of Multifetal Pregnancies","volume":"335","author":"Wapner","year":"1990","journal-title":"Lancet"},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"3130","DOI":"10.1093\/oxfordjournals.humrep.a135872","article-title":"Sequential Genetic Events Leading to Conjoined Twins in a Monozygotic Triplet Pregnancy","volume":"10","author":"Lipitz","year":"1995","journal-title":"Hum. Reprod."},{"key":"ref_21","first-page":"235873","article-title":"Conjoined Twins in a Triplet Pregnancy: A Case Report","volume":"2011","author":"Shepherd","year":"2011","journal-title":"Case Rep. Obstet. Gynecol."},{"key":"ref_22","first-page":"351","article-title":"Conjoined Twins in a Spontaneous Trichorionic Quadruplet Pregnancy: A Case Report","volume":"56","author":"Rohilla","year":"2011","journal-title":"J. Reprod. Med."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"353","DOI":"10.1007\/s00247-009-1445-9","article-title":"Fetal MRI of Conjoined Twins Who Switched Their Relative Positions","volume":"40","author":"Huisman","year":"2009","journal-title":"Pediatr. Radiol."},{"key":"ref_24","first-page":"552","article-title":"Early Diagnosis of Conjoined Twins Using Two-Dimensional Color Doppler and Three-Dimensional Ultrasound","volume":"90","author":"Raga","year":"1998","journal-title":"J. Natl. Med. Assoc."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"145","DOI":"10.1097\/01.AOG.0000318871.95090.d9","article-title":"Prenatal Imaging: Ultrasonography and Magnetic Resonance Imaging","volume":"112","author":"Reddy","year":"2008","journal-title":"Obstet. Gynecol."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"510","DOI":"10.1016\/S0015-0282(16)56169-X","article-title":"Increased Risk of Monochorionic Twinning Associated with Assisted Reproduction","volume":"60","author":"Wenstrom","year":"1993","journal-title":"Fertil. Steril."},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"268.e7","DOI":"10.1016\/j.fertnstert.2009.08.054","article-title":"Conjoined Twins after Intracytoplasmic Sperm Injection and Transfer of a Single Day 2 Embryo: Case Report","volume":"93","author":"Poret","year":"2010","journal-title":"Fertil. Steril."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"557","DOI":"10.1016\/S0002-9378(89)80025-0","article-title":"The Ultrasonographic Prediction of Chorionicity in Twin Gestation","volume":"160","author":"Dudley","year":"1989","journal-title":"Am. J. Obstet. Gynecol."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1016\/0029-7844(96)00094-4","article-title":"Evaluation of the Twin Peak or Lambda Sign in Determining Chorionicity in Multiple Pregnancy","volume":"88","author":"Wood","year":"1996","journal-title":"Obstet. Gynecol."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"503","DOI":"10.1016\/S0015-0282(02)04754-4","article-title":"Incidence of Monozygotic Twinning with Blastocyst Transfer Compared to Cleavage-Stage Transfer","volume":"79","author":"Milki","year":"2003","journal-title":"Fertil. Steril."},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"217","DOI":"10.1016\/S0015-0282(00)01635-6","article-title":"The Potential Association between Blastocyst Transfer and Monozygotic Twinning","volume":"75","author":"Sheiner","year":"2001","journal-title":"Fertil. Steril."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"933.e9","DOI":"10.1016\/j.fertnstert.2008.07.1730","article-title":"Conjoined Twins in a Triplet Pregnancy after Intracytoplasmic Sperm Injection and Blastocyst Transfer: Case Report and Review of the Literature","volume":"91","author":"Hirata","year":"2009","journal-title":"Fertil. Steril."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"144","DOI":"10.1016\/S0015-0282(98)00402-6","article-title":"Monozygotic Twinning Associated with Mechanical Assisted Hatching","volume":"71","author":"Hershlag","year":"1999","journal-title":"Fertil. Steril."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"396","DOI":"10.1093\/humupd\/6.4.396","article-title":"Monozygotic Twinning and IVF\/ICSI Treatment: A Report of 11 Cases and Review of Literature","volume":"6","author":"Abusheika","year":"2000","journal-title":"Hum. Reprod. Update"},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"196","DOI":"10.1016\/S0015-0282(01)02958-2","article-title":"Increase in the Monozygotic Twinning Rate after Intracytoplasmic Sperm Injection and Blastocyst Stage Embryo Transfer","volume":"77","author":"Tarlatzis","year":"2002","journal-title":"Fertil. Steril."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"114","DOI":"10.1007\/BF01139357","article-title":"Identical Twins and in Vitro Fertilization","volume":"3","author":"Edwards","year":"1986","journal-title":"J. In Vitro Fert. Embryo Transf."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1093\/oxfordjournals.humrep.a137044","article-title":"Impairment of the Hatching Process following IVF in the Human and Improvement of Implantation by Assisting Hatching Using Micromanipulation","volume":"5","author":"Cohen","year":"1990","journal-title":"Hum. Reprod."},{"key":"ref_38","doi-asserted-by":"crossref","first-page":"e111","DOI":"10.1016\/j.fertnstert.2011.06.002","article-title":"Conjoined Twins after Intracytoplasmic Sperm Injection and Transfer of Day-3 Embryos","volume":"96","author":"Mercan","year":"2011","journal-title":"Fertil. Steril."},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"239","DOI":"10.1002\/(SICI)1096-8628(19971219)73:3<239::AID-AJMG1>3.0.CO;2-N","article-title":"Heteropagus Conjoined Twins Due to Fusion of Two Embryos: Report and Review\u2014PubMed","volume":"73","author":"Harris","year":"1997","journal-title":"Am. J. Med. Genet."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"611","DOI":"10.7863\/jum.1995.14.8.611","article-title":"Early Diagnosis of Conjoined Twins in Triplet Pregnancy after in Vitro Fertilization and Assisted Hatching","volume":"14","author":"Skupski","year":"1995","journal-title":"J. Ultrasound Med."},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"669","DOI":"10.1046\/j.1469-0705.2000.00281.x","article-title":"Dichorionic Triplet Pregnancy with the Monoamniotic Twin Pair Concordant for Omphalocele and Bladder Exstrophy","volume":"16","author":"Monteagudo","year":"2000","journal-title":"Ultrasound Obstet. Gynecol."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"666","DOI":"10.1007\/s10024-005-0009-8","article-title":"Diamniotic Conjoined Fetuses in a Triplet Pregnancy: An Insight into Embryonic Topology: Pediatr","volume":"8","author":"Charles","year":"2005","journal-title":"Dev. Pathol."},{"key":"ref_43","first-page":"729","article-title":"Conjoined Twins in a Monochorionic Triplet Pregnancy after in Vitro Fertilization: A Case Report","volume":"13","author":"Talebian","year":"2015","journal-title":"Iran. J. Reprod. Med."},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"85","DOI":"10.1007\/s40477-016-0235-0","article-title":"First-Trimester Diagnosis of Conjoined Twins in a Multifetal Pregnancy after Assisted Reproduction Technique Using HDlive Rendering","volume":"20","author":"Castro","year":"2017","journal-title":"J. Ultrasound"},{"key":"ref_45","doi-asserted-by":"crossref","first-page":"196","DOI":"10.1002\/ijgo.12123","article-title":"Triplet Pregnancy from the Transfer of Two Blastocysts Demonstrating a Twin Reversed Arterial Perfusion Sequence with a Conjoined-Twins Pump Fetus","volume":"137","author":"Yuan","year":"2017","journal-title":"Int. J. Gynecol. Obstet."},{"key":"ref_46","doi-asserted-by":"crossref","unstructured":"Liu, H., Deng, C., Hu, Q., Liao, H., Wang, X., and Yu, H. (2021). Conjoined Twins in Dichorionic Diamniotic Triplet Pregnancy: A Report of Three Cases and Literature Review. BMC Pregnancy Childbirth, 21.","DOI":"10.1186\/s12884-021-04165-x"}],"container-title":["Children"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2227-9067\/9\/10\/1549\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T00:52:59Z","timestamp":1760143979000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2227-9067\/9\/10\/1549"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,10,12]]},"references-count":46,"journal-issue":{"issue":"10","published-online":{"date-parts":[[2022,10]]}},"alternative-id":["children9101549"],"URL":"https:\/\/doi.org\/10.3390\/children9101549","relation":{},"ISSN":["2227-9067"],"issn-type":[{"value":"2227-9067","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,10,12]]}}}