{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,29]],"date-time":"2026-05-29T19:42:19Z","timestamp":1780083739603,"version":"3.54.0"},"reference-count":10,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2022,3,29]],"date-time":"2022-03-29T00:00:00Z","timestamp":1648512000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2022,3,29]],"date-time":"2022-03-29T00:00:00Z","timestamp":1648512000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Endocr Disord"],"published-print":{"date-parts":[[2022,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Vulvar neuroendocrine carcinomas with small cell morphology need an appropriate differential diagnosis with respect to primary Merkel cell carcinomas, primary small cell neuroendocrine carcinomas, and secondary\/metastatic carcinomas. Herein, we report a woman with a history of endometrial carcinoma led to neuroendocrine vulvar carcinoma.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>An 82-y-old woman with right vulvar swelling was transferred to our hospital. Computed tomography scan showed a 75\u00a0mm irregular mass in her right vulva. Three years ago, she had been diagnosed with endometrial endometrioid carcinoma stage IA and had undergone surgery. Vulvar biopsy revealed neuroendocrine carcinomas with small cell morphology. Immunohistochemical staining showed that the vulvar tumor was positive for CD56 and chromogranin A, but negative for Merkel cell polyomavirus and cytokeratin 20. Incidentally, her endometrial carcinoma was also positive for CD56 and chromogranin A. Human papillomavirus DNA typing analysis of vulvar tumor was negative. Hence, the vulvar tumor seemed to be a recurrence of the endometrial cancer rather than a primary vulvar neuroendocrine carcinoma. The patient died of the disease within a month.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>We report a case of vulvar neuroendocrine carcinoma that is independent of Merkel cell polyomavirus and human papillomavirus, thereby suggesting a recurrence of endometrial cancer. Immunohistochemical and virological analyses helped in the differential diagnosis of the neuroendocrine carcinoma.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12902-022-00987-8","type":"journal-article","created":{"date-parts":[[2022,3,29]],"date-time":"2022-03-29T10:14:58Z","timestamp":1648548898000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Vulvar neuroendocrine carcinoma that is independent of merkel cell polyomavirus and human papillomavirus suggests endometrial cancer recurrence: a case report"],"prefix":"10.1186","volume":"22","author":[{"given":"Tomoko","family":"Hirakawa","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4436-838X","authenticated-orcid":false,"given":"Mitsutake","family":"Yano","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haruto","family":"Nishida","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shimpei","family":"Sato","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaei","family":"Nasu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2022,3,29]]},"reference":[{"key":"987_CR1","doi-asserted-by":"publisher","first-page":"304","DOI":"10.1097\/PAS.0000000000001558","volume":"45","author":"PP Chen","year":"2021","unstructured":"Chen PP, Ramalingam P, Alvarado-Cabrero I, Euscher ED, Nagarajan P, Lawson BC, et al. High-grade neuroendocrine carcinomas of the vulva: a clinicopathologic study of 16 cases. Am J Surg Pathol. 2021;45:304\u201316.","journal-title":"Am J Surg Pathol"},{"key":"987_CR2","doi-asserted-by":"publisher","first-page":"577","DOI":"10.1097\/PAS.0000000000000633","volume":"40","author":"CE Pocrnich","year":"2016","unstructured":"Pocrnich CE, Ramalingam P, Euscher ED, Malpica A. Neuroendocrine carcinoma of the endometrium: a clinicopathologic study of 25 cases. Am J Surg Pathol. 2016;40:577\u201386.","journal-title":"Am J Surg Pathol"},{"key":"987_CR3","doi-asserted-by":"publisher","first-page":"654","DOI":"10.1016\/j.prp.2016.03.009","volume":"212","author":"K Ono","year":"2016","unstructured":"Ono K, Yokota NR, Yoshioka E, Noguchi A, Washimi K, Kawachi K, et al. Metastatic large cell neuroendocrine carcinoma of the lung arising from the uterus: a pitfall in lung cancer diagnosis. Pathol Res Pract. 2016;212:654\u20137.","journal-title":"Pathol Res Pract"},{"issue":"5","key":"987_CR4","first-page":"114","volume":"2016","author":"A D'Antonio","year":"2012","unstructured":"D\u2019Antonio A, Addesso M, Caleo A, Guida M, Zeppa P. Small cell neuroendocrine carcinoma of the endometrium with pulmonary metastasis: a clinicopathologic study of a case and a brief review of the literature. Annals of medicine and surgery. 2012;2016(5):114\u20137.","journal-title":"Annals of medicine and surgery"},{"issue":"1","key":"987_CR5","doi-asserted-by":"publisher","first-page":"35","DOI":"10.1097\/PGP.0000000000000770","volume":"41","author":"VI Shah","year":"2022","unstructured":"Shah VI, Morgan SE, K\u00f6bel M, Lee CH, McCluggage WG. Dedifferentiation in Breast Metastasis of Endometrial Carcinoma: A Diagnostic Dilemma. Int J Gynecol Pathol. 2022;41(1):35\u20139.","journal-title":"Int J Gynecol Pathol."},{"key":"987_CR6","doi-asserted-by":"publisher","first-page":"821","DOI":"10.31083\/j.ejgo.2020.05.5429","volume":"41","author":"YH Ikuji Shimazaki","year":"2020","unstructured":"Ikuji Shimazaki YH, Yamauchi M, Kasai M, Fukuda T, Ichimura T, Yasui T, Sumi T. Secondary small cell vaginal cancer after operative therapy for endometrial cancer. Secondary Eur J Gynaecol Oncol. 2020;41:821\u20133.","journal-title":"Secondary. Eur J Gynaecol Oncol."},{"issue":"7","key":"987_CR7","doi-asserted-by":"publisher","first-page":"1023","DOI":"10.1038\/s41379-019-0220-1","volume":"32","author":"M Yano","year":"2019","unstructured":"Yano M, Ito K, Yabuno A, Ogane N, Katoh T, Miyazawa M, et al. Impact of TP53 immunohistochemistry on the histological grading system for endometrial endometrioid carcinoma. Mod Pathol. 2019;32(7):1023\u201331.","journal-title":"Mod Pathol."},{"key":"987_CR8","doi-asserted-by":"publisher","first-page":"e25863","DOI":"10.1097\/MD.0000000000025863","volume":"100","author":"VD Mandato","year":"2021","unstructured":"Mandato VD, Mastrofilippo V, Palicelli A, Silvotti M, Serra S, Giaccherini L, et al. Solitary vulvar metastasis from early-stage endometrial cancer: Case report and literature review. Med. 2021;100.","journal-title":"Med."},{"key":"987_CR9","doi-asserted-by":"crossref","first-page":"2050313x2199920","DOI":"10.1177\/2050313X21999200","volume":"9","author":"K Inoue","year":"2021","unstructured":"Inoue K, Kai K, Sato S, Nishida H, Hirakawa K, Nasu K, et al. Mixed large and small cell neuroendocrine carcinoma and endometrioid carcinoma of the endometrium with high microsatellite instability: a case report and literature review. SAGE Open Med Case Rep. 2021;9:2050313x21999200.","journal-title":"SAGE Open Med Case Rep"},{"key":"987_CR10","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1200\/JCO.19.02105","volume":"38","author":"A Marabelle","year":"2020","unstructured":"Marabelle A, Le DT, Ascierto PA, Di Giacomo AM, De Jesus-Acosta A, Delord JP, et al. Efficacy of Pembrolizumab in Patients With Noncolorectal High Microsatellite Instability\/Mismatch Repair-Deficient Cancer: Results From the Phase II KEYNOTE-158 Study. J Clin Oncol. 2020;38:1\u201310.","journal-title":"J Clin Oncol"}],"container-title":["BMC Endocrine Disorders"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-022-00987-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12902-022-00987-8\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-022-00987-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,3,29]],"date-time":"2022-03-29T10:15:40Z","timestamp":1648548940000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcendocrdisord.biomedcentral.com\/articles\/10.1186\/s12902-022-00987-8"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,3,29]]},"references-count":10,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2022,12]]}},"alternative-id":["987"],"URL":"https:\/\/doi.org\/10.1186\/s12902-022-00987-8","relation":{},"ISSN":["1472-6823"],"issn-type":[{"value":"1472-6823","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,3,29]]},"assertion":[{"value":"25 November 2021","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"13 March 2022","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"29 March 2022","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient and the next-of-kin for the publication of this case report (including clinical details and images).","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare no conflicts of interest associated with this manuscript.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"81"}}