{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,23]],"date-time":"2025-10-23T11:21:19Z","timestamp":1761218479115,"version":"build-2065373602"},"reference-count":39,"publisher":"MDPI AG","issue":"9","license":[{"start":{"date-parts":[[2022,4,20]],"date-time":"2022-04-20T00:00:00Z","timestamp":1650412800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJERPH"],"abstract":"<jats:p>Background: The aim of the present study is to report a rare occurrence of a successful twin pregnancy in a woman with pure 46,XY gonadal dysgenesis. Result(s): A patient with Swyer syndrome (pure 46,XY gonadal dysgenesis) presented with a twin pregnancy after in vitro fertilization. Due to unidentified conditions, the patient developed selective intrauterine growth restriction in one of the fetuses. Twins were born at 33 weeks of pregnancy due to the risk of asphyxia. Nonetheless, the patient did not develop gonadal malignancies before the pregnancy and, despite receiving estrogen, remained amenorrheic. Conclusion(s): The aim of this case report is to show the course of twin pregnancy in patients with Swyer syndrome through assisted reproduction. Due to certain disorders in the development of their reproductive organs, such as the less mature uterus, such pregnancies may be associated with an increased risk. The above case report demonstrates the need to systematize methods of pregnancy management in patients with Swyer syndrome, such as: preparation for the pregnancy, assessment of the uterus, medications used, and necessary checkups. Capsule: This case report and review shows clinicians that patients with Swyer syndrome may become pregnant. Twin pregnancies may occur without any major problems through assisted reproduction.<\/jats:p>","DOI":"10.3390\/ijerph19095027","type":"journal-article","created":{"date-parts":[[2022,4,21]],"date-time":"2022-04-21T01:55:51Z","timestamp":1650506151000},"page":"5027","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["A Successful New Case of Twin Pregnancy in a Patient with Swyer Syndrome\u2014An Up-to-Date Review on the Incidence and Outcome of Twin\/Multiple Gestations in the Pure 46,XY Gonadal Dysgenesis"],"prefix":"10.3390","volume":"19","author":[{"given":"Izabela","family":"Winkler","sequence":"first","affiliation":[{"name":"Second Department of Gynecological Oncology, St. John\u2019s Center of Oncology of the Lublin Region, 7 Jaczewskiego Street, 20-090 Lublin, Poland"},{"name":"Second Department of Gynecology, Medical University of Lublin, 20-954 Lublin, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5551-6915","authenticated-orcid":false,"given":"Ilona","family":"Jaszczuk","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology, Oncology and Transplantology, Children Clinical Hospital, II Department of Pediatrics, Medical University of Lublin, 20-093 Lublin, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9630-2086","authenticated-orcid":false,"given":"Marek","family":"Gogacz","sequence":"additional","affiliation":[{"name":"Second Department of Gynecology, Medical University of Lublin, 20-954 Lublin, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3582-067X","authenticated-orcid":false,"given":"Piotr","family":"Szkodziak","sequence":"additional","affiliation":[{"name":"Third Department of Gynecology, Medical University of Lublin, 20-954 Lublin, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0256-9853","authenticated-orcid":false,"given":"Tomasz","family":"Paszkowski","sequence":"additional","affiliation":[{"name":"Third Department of Gynecology, Medical University of Lublin, 20-954 Lublin, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Katarzyna","family":"Skorupska","sequence":"additional","affiliation":[{"name":"Second Department of Gynecology, Medical University of Lublin, 20-954 Lublin, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5780-5983","authenticated-orcid":false,"given":"Micha\u0142","family":"Ciebiera","sequence":"additional","affiliation":[{"name":"Second Department of Obstetrics and Gynecology, Center of Postgraduate Medical Education, 01-809 Warsaw, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maciej","family":"Skrzypczak","sequence":"additional","affiliation":[{"name":"Second Department of Gynecology, Medical University of Lublin, 20-954 Lublin, Poland"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2022,4,20]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"709","DOI":"10.1136\/bmj.2.4941.709","article-title":"Male pseudohermaphroditism: A hitherto undescribed form","volume":"2","author":"Swyer","year":"1955","journal-title":"Br. Med. J."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"737","DOI":"10.1111\/j.1471-0528.2008.01703.x","article-title":"Swyer syndrome: Presentation and outcomes","volume":"115","author":"Michala","year":"2008","journal-title":"BJOG Int. J. Obstet. Gynaecol."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"452","DOI":"10.1038\/348452a0","article-title":"A human XY female with a frame shift mutation in the candidate testis-determining gene SRY","volume":"348","author":"Jager","year":"1990","journal-title":"Nature"},{"key":"ref_4","unstructured":"(2021, October 21). Available online: https:\/\/www.uptodate.com\/contents\/causes-of-primary-amenorrhea."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"13","DOI":"10.1016\/j.ijgo.2015.06.020","article-title":"FIGO consensus guidelines on intrapartum fetal monitoring: Cardiotocography","volume":"131","author":"Spong","year":"2015","journal-title":"Int. J. Gynecol. Obstet."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"44","DOI":"10.1002\/uog.19073","article-title":"Fetal Medicine Foundation fetal and neonatal population weight charts","volume":"52","author":"Nicolaides","year":"2021","journal-title":"Ultrasound. Obstet. Gynecol."},{"key":"ref_7","unstructured":"Fetal Medicine Foundation (2021, October 21). The Fetal Medicine Foundation. Accredit 11-13+6 Week Scan., Available online: https:\/\/fetalmedicine.org\/research\/assess\/growth."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"603","DOI":"10.1038\/nrendo.2014.130","article-title":"DSDs: Genetics, underlying pathologies and psychosexual differentiation","volume":"10","author":"Arboleda","year":"2014","journal-title":"Nat. Rev. Endocrinol."},{"key":"ref_9","first-page":"E958","article-title":"Patients with disorders of sex development (DSD) at risk of gonadal tumour development: Management based on laparoscopic biopsy and molecular diagnosis","volume":"110","author":"Holterhus","year":"2012","journal-title":"BJU Int."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1431","DOI":"10.1016\/j.fertnstert.2011.09.009","article-title":"Complete gonadal dysgenesis in clinical practice: The 46,XY karyotype accounts for more than one third of cases","volume":"96","author":"Rocha","year":"2011","journal-title":"Fertil. Steril."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"621","DOI":"10.1111\/cen.13255","article-title":"Gonadal tumour risk in 292 phenotypic female patients with disorders of sex development containing Y chromosome or Y-derived sequence","volume":"86","author":"Huang","year":"2017","journal-title":"Clin. Endocrinol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"368","DOI":"10.1016\/j.jpurol.2012.12.002","article-title":"Review and management of 46,XY disorders of sex development","volume":"9","author":"Massanyi","year":"2013","journal-title":"J. Pediatr. Urol."},{"key":"ref_13","unstructured":"(2021, October 21). Available online: https:\/\/www.eshre.eu\/-\/media\/sitecore-files\/Guidelines\/POI\/ESHRE-guideline_POI-2015_summary_01122015.pdf?la=en&hash=04AFBA15E89FAAD0D81BB41DD64403BE6B04678E."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"645","DOI":"10.1016\/j.jpag.2019.07.007","article-title":"Atypical Presentation of Swyer Syndrome","volume":"32","author":"Dural","year":"2019","journal-title":"J. Pediatr. Adolesc. Gynecol."},{"key":"ref_15","first-page":"792","article-title":"La dysgenesie gonadidique pure a 46 XY","volume":"25","author":"Coutin","year":"1996","journal-title":"J. Gynecol. Obstet. Biol. Reprod."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"217","DOI":"10.1016\/j.fertnstert.2004.11.087","article-title":"Successful pregnancy in a gonadectomised woman with 46, XY gonadal dysgenesis and gonadoblastoma","volume":"84","author":"Chen","year":"2005","journal-title":"Fertil. Steril."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"704","DOI":"10.1093\/humupd\/dmq017","article-title":"Clinicaland immunologic aspects of oocyte donation pregnancies: A systematic review","volume":"16","author":"Lashley","year":"2010","journal-title":"Hum. Reprod. Update"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"620","DOI":"10.1093\/humupd\/dmw012","article-title":"Maternal and fetal outcomes in oocyte donation pregnancies","volume":"22","author":"Savasi","year":"2016","journal-title":"Hum. Reprod. Update"},{"key":"ref_19","first-page":"1","article-title":"Rare successful pregnancy in a patient with Swyer Syndrome","volume":"12","author":"Taneja","year":"2014","journal-title":"Case Rep. Women\u2019s Health"},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"380","DOI":"10.1016\/0002-9378(89)90525-5","article-title":"Successful twin pregnancy after embryo donation to a patient with XY gonadal dysgenesis","volume":"161","author":"Sauer","year":"1989","journal-title":"Am. J. Obstet. Gynecol."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"1434","DOI":"10.1093\/humrep\/12.7.1434","article-title":"Two successful pregnancies in a 46, XY patient","volume":"12","author":"Kan","year":"1997","journal-title":"Hum. Reprod."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"276","DOI":"10.1016\/S1028-4559(08)60034-0","article-title":"Triplet pregnancy complicated with one hydatidiform mole and preeclampsia in a 46, XY female with gonadal dysgenesis","volume":"46","author":"Ko","year":"2007","journal-title":"Taiwan J. Obstet. Gynecol."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"1789","DOI":"10.1016\/j.fertnstert.2010.12.012","article-title":"Pregnancy in patient with Swyer syndrome","volume":"95","author":"Tulic","year":"2011","journal-title":"Fertil. Steril."},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"e83","DOI":"10.1016\/j.fertnstert.2011.05.061","article-title":"Successful pregnancy in a Swyer syndrome patient with preexisting hypertension","volume":"96","author":"Creatsas","year":"2011","journal-title":"Fertil. Steril."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"419","DOI":"10.1016\/S0015-0282(02)03242-9","article-title":"Successful pregnancy in a patient with a 46,XY karyotype","volume":"78","author":"Selvaraj","year":"2002","journal-title":"Fertil. Steril."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"174","DOI":"10.1038\/307174a0","article-title":"The establishment and maintenance of pregnancy using in vitro fertilization and embryo donation in a patient with primary ovarian failure","volume":"307","author":"Lutjen","year":"1984","journal-title":"Nature"},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"291","DOI":"10.1093\/oxfordjournals.humrep.a137092","article-title":"Pregnancies following ovum donation in gonadal dysgenesis","volume":"5","author":"Cornet","year":"1990","journal-title":"Hum. Reprod."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"101","DOI":"10.1016\/S0301-2115(99)00110-4","article-title":"Subsequent successful pregnancy and delivery after intracytoplasmic sperm injection in a patient with XY gonadal dysgenesisms","volume":"88","author":"Dirnfeld","year":"2000","journal-title":"Eur. J Obstet. Gynecol. Reprod. Biol."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"2015.e1","DOI":"10.1016\/j.fertnstert.2008.04.043","article-title":"A case report of successful pregnancy in a patient with pure 46, XY gonadal dysgenesis","volume":"90","author":"Plante","year":"2008","journal-title":"Fertil. Steril."},{"key":"ref_30","first-page":"502","article-title":"Multifetal pregnancy in a gonadal dysgenesis mosaic","volume":"76","author":"Bardeguez","year":"1990","journal-title":"Obstet. Gynecol."},{"key":"ref_31","first-page":"37","article-title":"Successful pregnancy in a pure gonadal dysgenesis with karyotype 46,XY patient (Swyer\u2019s syndrome) following oocyte donation and hormonal treatment","volume":"23","author":"Bianco","year":"1992","journal-title":"Acta Eur. Fertil."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"813","DOI":"10.1016\/S0015-0282(16)60322-9","article-title":"Pregnancy in a 46, XY patient","volume":"50","author":"Frydman","year":"1988","journal-title":"Fertil. Steril."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"737","DOI":"10.3109\/01443615.2013.822480","article-title":"Successful twin pregnancy in a 46,XY pure gonadal dysgenesis","volume":"33","author":"Spagnuolo","year":"2013","journal-title":"J. Obstet. Gynaecol."},{"key":"ref_34","first-page":"1109","article-title":"Twin delivery of a 46,XY gonadal dysgenetic woman following vitrified oocytes donation","volume":"124","author":"Gao","year":"2011","journal-title":"Chin. Med. J. Engl."},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"1425","DOI":"10.1007\/s00404-013-2933-6","article-title":"Unexpected labor and successful twin birth to a pure gonadal dysgenetic woman","volume":"288","author":"Murtinger","year":"2013","journal-title":"Arch. Gynecol. Obstet."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"2061","DOI":"10.18203\/2320-1770.ijrcog20161722","article-title":"Successful pregnancy outcome in a case of Swyer Syndrome with hypertension and morbid obesity","volume":"5","author":"Kalra","year":"2016","journal-title":"Int. J. Reprod. Contracept. Obstet. Gynecol."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"719","DOI":"10.1080\/01443615.2017.1380615","article-title":"Twin gestation in a Swyer syndrome patient with superimposed pre-eclampsia","volume":"38","author":"Shah","year":"2018","journal-title":"J. Obstet. Gynecol."},{"key":"ref_38","first-page":"158","article-title":"Two successful pregnancies after in vitro fertilisation with oocyte donation in a patient with Swyer syndrome\u2014A case report","volume":"20","author":"Urban","year":"2021","journal-title":"Prz. Menopauzalny"},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"267","DOI":"10.4103\/jhrs.JHRS_14_19","article-title":"A rare case of Swyer syndrome in two sisters with successful pregnancy outcome in both","volume":"12","author":"Gupta","year":"2019","journal-title":"J. Hum. Reprod. Sci."}],"container-title":["International Journal of Environmental Research and Public Health"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/1660-4601\/19\/9\/5027\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T22:57:49Z","timestamp":1760137069000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/1660-4601\/19\/9\/5027"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,4,20]]},"references-count":39,"journal-issue":{"issue":"9","published-online":{"date-parts":[[2022,5]]}},"alternative-id":["ijerph19095027"],"URL":"https:\/\/doi.org\/10.3390\/ijerph19095027","relation":{},"ISSN":["1660-4601"],"issn-type":[{"type":"electronic","value":"1660-4601"}],"subject":[],"published":{"date-parts":[[2022,4,20]]}}}