{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,3]],"date-time":"2026-02-03T16:37:52Z","timestamp":1770136672517,"version":"3.49.0"},"reference-count":20,"publisher":"MDPI AG","issue":"6","license":[{"start":{"date-parts":[[2021,12,13]],"date-time":"2021-12-13T00:00:00Z","timestamp":1639353600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000921","name":"European Cooperation in Science and Technology","doi-asserted-by":"publisher","award":["CA18117"],"award-info":[{"award-number":["CA18117"]}],"id":[{"id":"10.13039\/501100000921","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Current Oncology"],"abstract":"<jats:p>Epithelioid trophoblastic tumours are rare neoplasms showing differentiation towards the chorion leave-type intermediate cytotrophoblast, with only a handful of cases being reported in the literature. These tumours are slow-growing and are typically confined to the uterus for extended periods of time. While the pathogenesis is unclear, they are thought to arise from a remnant intermediate trophoblast originating from prior normal pregnancies or, less frequently, gestational trophoblastic tumours. A protracted time period between the gestational event and tumour development is typical. This case describes a 49-year-old previously healthy female who presented with a completely asymptomatic uterine mass, discovered incidentally during a routine gynaecological assessment. The pathological analysis of the hysterectomy specimen confirmed an epithelioid trophoblastic tumour, involving the uterus and cervix. This is a rare gynaecological tumour. A comparative short tandem repeat analysis revealed genetic similarities to a previous healthy gestation seventeen years prior. She was successful treated with adjuvant pembrolizumab, with no evidence of disease recurrence to date.<\/jats:p>","DOI":"10.3390\/curroncol28060446","type":"journal-article","created":{"date-parts":[[2021,12,14]],"date-time":"2021-12-14T01:20:41Z","timestamp":1639444841000},"page":"5346-5355","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["Epithelioid Trophoblastic Tumour: A Case with Genetic Linkage to a Child Born over Seventeen Years Prior, Successfully Treated with Surgery and Pembrolizumab"],"prefix":"10.3390","volume":"28","author":[{"given":"David","family":"Pisani","sequence":"first","affiliation":[{"name":"Department of Histopathology, Mater Dei Hospital, MSD2090 Msida, Malta"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6369-0297","authenticated-orcid":false,"given":"Jean","family":"Calleja-Agius","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Faculty of Medicine and Surgery, University of Malta, MSD2080 Msida, Malta"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4905-6948","authenticated-orcid":false,"given":"Riccardo","family":"Di Fiore","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Faculty of Medicine and Surgery, University of Malta, MSD2080 Msida, Malta"},{"name":"Sbarro Institute for Cancer Research and Molecular Medicine, Center for Biotechnology, College of Science and Technology, Temple University, Philadelphia, PA 19122, USA"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7311-9326","authenticated-orcid":false,"given":"John J.","family":"O\u2019Leary","sequence":"additional","affiliation":[{"name":"Department of Histopathology, Trinity College Dublin, Trinity St. James\u2019s Cancer Institute, St. James Hospital, D08 NHY1 Dublin, Ireland"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3053-3556","authenticated-orcid":false,"given":"James P.","family":"Beirne","sequence":"additional","affiliation":[{"name":"Department of Gynaecological Oncology, Trinity St James\u2019s Cancer Institute, St. James Hospital, D08 NHY1 Dublin, Ireland"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9260-3225","authenticated-orcid":false,"given":"Sharon A.","family":"O\u2019Toole","sequence":"additional","affiliation":[{"name":"Departments of Obstetrics and Gynaecology and Histopathology, Trinity St James\u2019s Cancer Institute, Trinity College Dublin, D08 NHY1 Dublin, Ireland"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2653-2262","authenticated-orcid":false,"given":"Ana","family":"Felix","sequence":"additional","affiliation":[{"name":"Department of Pathology, Campo dos M\u00e1rtires da P\u00e1tria, Instituto Portugues de Oncologia de Lisboa, NOVA Medical School, UNL, 130, 1169-056 Lisbon, Portugal"}]},{"given":"Ian","family":"Said-Huntingford","sequence":"additional","affiliation":[{"name":"Department of Histopathology, Mater Dei Hospital, MSD2090 Msida, Malta"}]}],"member":"1968","published-online":{"date-parts":[[2021,12,13]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"642","DOI":"10.1016\/S1470-2045(07)70204-8","article-title":"Gestational trophoblastic neoplasia-pathogenesis and potential therapeutic targets","volume":"8","author":"Shih","year":"2007","journal-title":"Lancet Oncol."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"3479","DOI":"10.1007\/s00018-019-03104-6","article-title":"Human placenta and trophoblast development: Key molecular mechanisms and model systems","volume":"76","author":"Haider","year":"2019","journal-title":"Cell Mol. Life Sci."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1016\/j.aanat.2015.10.003","article-title":"Intermediate trophoblast\u2014A distinctive, unique and often unrecognized population of trophoblastic cells","volume":"204","author":"Cierna","year":"2016","journal-title":"Ann. Anat. Anat. Anz."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"1393","DOI":"10.1097\/00000478-199811000-00010","article-title":"Epithelioid trophoblastic tumor: A neoplasm distinct from choriocarcinoma and placental site tropho-blastic tumor simulating carcinoma","volume":"22","author":"Shih","year":"1998","journal-title":"Am. J. Surg. Pathol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"684","DOI":"10.1016\/j.ygyno.2019.03.011","article-title":"Placental site trophoblastic tumor and epithelioid trophoblastic tumor: Clinical and pathological features, prognostic variables and treatment strategy","volume":"153","author":"Gadducci","year":"2019","journal-title":"Gynecol. Oncol."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"208","DOI":"10.1016\/j.ygyno.2016.10.024","article-title":"Placental site trophoblastic tumors and epithelioid trophoblastic tumors: Biology, natural history, and treatment modalities","volume":"144","author":"Horowitz","year":"2017","journal-title":"Gynecol. Oncol."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"1334","DOI":"10.1097\/IGC.0b013e31829ea023","article-title":"Epithelioid Trophoblastic Tumor: An Outcome-Based Literature Review of 78 Reported Cases","volume":"23","author":"Zhang","year":"2013","journal-title":"Int. J. Gynecol. Cancer"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"vi39","DOI":"10.1093\/annonc\/mdt345","article-title":"Gestational trophoblastic disease: ESMO Clinical Practice Guidelines for diagnosis, treatment and follow-up","volume":"24","author":"Seckl","year":"2013","journal-title":"Ann Oncol."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"960","DOI":"10.1111\/jog.13593","article-title":"Laparoscopic diagnosis and treatment of an isolated epithelioid trophoblastic tumor in recto-uterine pouch","volume":"44","author":"Jiang","year":"2018","journal-title":"J. Obstet. Gynaecol. Res."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1304","DOI":"10.1097\/GME.0000000000000908","article-title":"Two cases of epithelioid trophoblastic tumors in postmenopausal women","volume":"24","author":"Saco","year":"2017","journal-title":"Menopause"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"1558","DOI":"10.1097\/00000478-200011000-00014","article-title":"Epithelioid trophoblastic tumor of the uterus in a postmenopausal woman: A case report and review of the literature","volume":"24","author":"Coulson","year":"2000","journal-title":"Am. J. Surg. Pathol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"75","DOI":"10.1038\/modpathol.3800485","article-title":"Epithelioid trophoblastic tumor: Clinicopathological features with an emphasis on uterine cervical involvement","volume":"19","author":"Fadare","year":"2006","journal-title":"Mod. Pathol."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"67","DOI":"10.4132\/KoreanJPathol.2013.47.1.67","article-title":"Epithelioid Trophoblastic Tumor: Clinicopathologic and Immunohistochemical Analysis of Three Cases","volume":"47","author":"Sung","year":"2013","journal-title":"Korean J. Pathol."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"1117","DOI":"10.1097\/IGC.0000000000001278","article-title":"Surgery and Platinum\/Etoposide-Based Chemotherapy for the Treatment of Epithelioid Trophoblastic Tumor","volume":"28","author":"Winder","year":"2018","journal-title":"Int. J. Gynecol. Cancer"},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"e0306","DOI":"10.1097\/MD.0000000000010306","article-title":"Metastatic epithelioid trophoblastic tumor of the lung: A case report","volume":"97","author":"Lei","year":"2018","journal-title":"Medicine"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"124","DOI":"10.5468\/ogs.2017.60.1.124","article-title":"Extrauterine epithelioid trophoblastic tumor in hysterectomized woman","volume":"60","author":"Kim","year":"2017","journal-title":"Obstet. Gynecol. Sci."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"11","DOI":"10.7150\/jca.28134","article-title":"Epithelioid Trophoblastic Tumors: Treatments, Outcomes, and Potential Therapeutic Targets","volume":"10","author":"Yang","year":"2019","journal-title":"J. Cancer"},{"key":"ref_18","unstructured":"(2021, October 25). NCCN Guideline Gestational Trophoblastic Neoplasia Version 2.2021; 31 Mar 21. Available online: https:\/\/www.nccn.org\/professionals\/physician_gls\/pdf\/gtn.pdf."},{"key":"ref_19","unstructured":"(2021, October 24). NHS Urgent Clinical Commissioning Policy Statement: Pembrolizumab for Drug-Resistant Gestational Trophoblastic Neo-plasia. NHS England Reference: 170027P. Available online: https:\/\/www.england.nhs.uk\/wp-content\/uploads\/2018\/12\/Pembrolizumab-for-drug-resistant-gestational-trophoblastic-neoplasia.pdf."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1016\/j.jogc.2020.03.001","article-title":"Guideline No. 408: Management of Gestational Trophoblastic Diseases","volume":"43","author":"Eiriksson","year":"2021","journal-title":"J. Obstet. Gynaecol. Can."}],"container-title":["Current Oncology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/1718-7729\/28\/6\/446\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T07:47:06Z","timestamp":1760168826000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/1718-7729\/28\/6\/446"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,12,13]]},"references-count":20,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2021,12]]}},"alternative-id":["curroncol28060446"],"URL":"https:\/\/doi.org\/10.3390\/curroncol28060446","relation":{},"ISSN":["1718-7729"],"issn-type":[{"value":"1718-7729","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,12,13]]}}}