{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2024,9,15]],"date-time":"2024-09-15T23:25:58Z","timestamp":1726442758617},"reference-count":15,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,7,7]],"date-time":"2021-07-07T00:00:00Z","timestamp":1625616000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2021,7,7]],"date-time":"2021-07-07T00:00:00Z","timestamp":1625616000000},"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":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Vaginal paraganglioma are rare, atypical, solitary tumors which originate from the female genital tract. Sacrococcygeal teratoma are also rare neoplasms which derive from one (or more) primordial germ cell layers. Here we report a unique case of vaginal paraganglioma with sacrococcygeal teratoma.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>A 44-year-old female experienced paroxysmal hypertension, palpitations and dizziness for almost six years. Enhanced CT and MRI highlighted two abnormal soft tissue lesions located in the left vaginal wall and coccyx anteriorly, and Iodine-131 metaiodobenzylguanidine (<jats:sup>131<\/jats:sup>I-MIBG) demonstrated abnormal radioactive uptake in perineum area. Endocrine tests showed elevated plasma normetanephrine (NMN) and 24\u00a0h urine norepinephrine. There was a well-circumscribed soft tissue lesion of approximately 3.5\u00a0cm in the left lateral vaginal wall which could be palpated during bimanual examination, together with a 1.5\u00a0cm tumor in the posterior wall of the rectum. We completely resected the two lesions in stages with the support of a senior gynecologist and general surgeons. Postoperative histopathological examinations suggested the vaginal paraganglioma and mature sacrococcygeal teratoma. Targeted sanger sequencing for the 36 mostly common paraganglioma-related genes, with a depth of 1000x, revealed no mutations. Post-operatively, plasma NMN and 24\u00a0h urine norepinephrine returned to the normal range and her symptoms completely disappeared.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>We reported an extremely rare case and the successful treatment of functional vaginal paraganglioma coexisting with adult sacrococcygeal teratoma.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12902-021-00806-6","type":"journal-article","created":{"date-parts":[[2021,7,7]],"date-time":"2021-07-07T07:03:14Z","timestamp":1625641394000},"update-policy":"http:\/\/dx.doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Solitary vaginal paraganglioma with mature sacrococcygeal teratoma: a rare case report"],"prefix":"10.1186","volume":"21","author":[{"given":"Zhan","family":"Wang","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hua","family":"Fan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jinghua","family":"Fan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Samuel","family":"Seery","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wenda","family":"Wang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yushi","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2021,7,7]]},"reference":[{"issue":"2","key":"806_CR1","doi-asserted-by":"publisher","first-page":"286","DOI":"10.1007\/s13193-017-0688-4","volume":"9","author":"S Sharma","year":"2018","unstructured":"Sharma S, Chougule A, Garg R, Chopra S, Sikka P. A Long-Standing Primary Vaginal Paraganglioma-Coexisting with Esophageal Carcinoma. Indian J Surg Oncol. 2018;9(2):286\u20137.","journal-title":"Indian J Surg Oncol"},{"issue":"8","key":"806_CR2","doi-asserted-by":"publisher","first-page":"670","DOI":"10.1631\/jzus.B1800621","volume":"20","author":"XM Xu","year":"2019","unstructured":"Xu XM, Zhao F, Cheng XF, Zhong WX, Liu JP, Jiang WQ, Yu XK, Lin JJ. Adult sacrococcygeal teratoma: a retrospective study over eight years at a single institution. J Zhejiang Univ Sci B. 2019;20(8):670\u20138.","journal-title":"J Zhejiang Univ Sci B"},{"issue":"1","key":"806_CR3","doi-asserted-by":"publisher","first-page":"144","DOI":"10.1186\/s40792-019-0702-x","volume":"5","author":"L Wang","year":"2019","unstructured":"Wang L, Hirano Y, Ishii T, Kondo H, Hara K, Ishikawa S, Okada T, Obara N, Yamaguchi S. Laparoscopic surgical management of a mature presacral teratoma: a case report. Surg Case Rep. 2019;5(1):144.","journal-title":"Surg Case Rep"},{"issue":"5","key":"806_CR4","doi-asserted-by":"publisher","first-page":"683","DOI":"10.1007\/s00268-007-9360-4","volume":"32","author":"JA Lee","year":"2008","unstructured":"Lee JA, Duh QY. Sporadic paraganglioma. World J Surg. 2008;32(5):683\u20137.","journal-title":"World J Surg"},{"issue":"6","key":"806_CR5","doi-asserted-by":"publisher","first-page":"599","DOI":"10.1097\/PGP.0000000000000658","volume":"39","author":"RW Wong","year":"2020","unstructured":"Wong RW, Liu APY, Choi CKM, Chan AOK. Paraganglioma of the Vagina Associated With Germline SDHB Mutation: report of a Case With Review of the Literature. Int J Gynecol Pathol. 2020;39(6):599\u2013604.","journal-title":"Int J Gynecol Pathol"},{"key":"806_CR6","doi-asserted-by":"publisher","first-page":"965","DOI":"10.2147\/OTT.S62174","volume":"7","author":"T Cai","year":"2014","unstructured":"Cai T, Li Y, Jiang Q, Wang D, Huang Y. Paraganglioma of the vagina: a case report and review of the literature. OncoTargets Ther. 2014;7:965\u20138.","journal-title":"OncoTargets Ther"},{"issue":"3","key":"806_CR7","doi-asserted-by":"publisher","first-page":"189","DOI":"10.1016\/j.prp.2007.01.002","volume":"203","author":"H Brustmann","year":"2007","unstructured":"Brustmann H. Paraganglioma of the vagina: report of a case. Pathol Res Pract. 2007;203(3):189\u201392.","journal-title":"Pathol Res Pract"},{"issue":"4","key":"806_CR8","doi-asserted-by":"publisher","first-page":"404","DOI":"10.1097\/01.pgp.0000092158.33490.24","volume":"22","author":"A Hassan","year":"2003","unstructured":"Hassan A, Bennet A, Bhalla S, Ylagan LR, Mutch D, Dehner LP. Paraganglioma of the vagina: report of a case, including immunohistochemical and ultrastructural findings. Int J Gynecol Pathol. 2003;22(4):404\u20136.","journal-title":"Int J Gynecol Pathol"},{"issue":"6","key":"806_CR9","doi-asserted-by":"publisher","first-page":"545","DOI":"10.3109\/08880019809018317","volume":"15","author":"SE Parkes","year":"1998","unstructured":"Parkes SE, Raafat F, Morland BJ. Paraganglioma of the vagina: the first report of a rare tumor in a child. Pediatr Hematol Oncol. 1998;15(6):545\u201351.","journal-title":"Pediatr Hematol Oncol"},{"issue":"1","key":"806_CR10","first-page":"35","volume":"139","author":"WP Plate","year":"1955","unstructured":"Plate WP. Phaeochromo-blastoma of the vagina. Gynaecol Int Monthly Rev Obstet Gynecol. 1955;139(1):35\u20139.","journal-title":"Gynaecol Int Monthly Rev Obstet Gynecol"},{"issue":"2","key":"806_CR11","doi-asserted-by":"publisher","first-page":"219","DOI":"10.1016\/0002-9378(81)90453-1","volume":"139","author":"G Pezeshkpour","year":"1981","unstructured":"Pezeshkpour G. Solitary paraganglioma of the vagina\u2013report of a case. Am J Obstetr Gynecol. 1981;139(2):219\u201321.","journal-title":"Am J Obstetr Gynecol"},{"issue":"2","key":"806_CR12","first-page":"184","volume":"29","author":"JG Shen","year":"2008","unstructured":"Shen JG, Chen YX, Xu DY, Feng YF, Tong ZH. Vaginal paraganglioma presenting as a gynecologic mass: case report. Eur J Gynaecol Oncol. 2008;29(2):184\u20135.","journal-title":"Eur J Gynaecol Oncol"},{"issue":"1","key":"806_CR13","doi-asserted-by":"publisher","first-page":"169","DOI":"10.1016\/j.surg.2008.08.020","volume":"147","author":"MN Akl","year":"2010","unstructured":"Akl MN, Naidu SG, McCullough AE, Magtibay PM. Vaginal paraganglioma presenting as a pelvic mass. Surgery. 2010;147(1):169\u201371.","journal-title":"Surgery"},{"key":"806_CR14","doi-asserted-by":"publisher","first-page":"117","DOI":"10.3389\/fonc.2014.00117","volume":"4","author":"N Matsumoto","year":"2014","unstructured":"Matsumoto N, Uehara K, Ando M, Arimoto J, Kato T, Nakamura H, Ebata T, Yokoyama Y, Nagino M. Adenocarcinoma arising from sacrococcygeal mature teratoma in an adult female: report of a case. Front Oncol. 2014;4:117.","journal-title":"Front Oncol"},{"issue":"5","key":"806_CR15","doi-asserted-by":"publisher","first-page":"977","DOI":"10.1007\/s00381-018-3718-9","volume":"34","author":"A Wessell","year":"2018","unstructured":"Wessell A, Hersh DS, Ho CY, Lumpkins KM, Groves MLA. Surgical treatment of a type IV cystic sacrococcygeal teratoma with intraspinal extension utilizing a posterior-anterior-posterior approach: a case report. Childs Nervous Syst. 2018;34(5):977\u201382.","journal-title":"Childs Nervous Syst"}],"container-title":["BMC Endocrine Disorders"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-021-00806-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12902-021-00806-6\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-021-00806-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,7,7]],"date-time":"2021-07-07T07:04:10Z","timestamp":1625641450000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcendocrdisord.biomedcentral.com\/articles\/10.1186\/s12902-021-00806-6"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,7,7]]},"references-count":15,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["806"],"URL":"https:\/\/doi.org\/10.1186\/s12902-021-00806-6","relation":{},"ISSN":["1472-6823"],"issn-type":[{"type":"electronic","value":"1472-6823"}],"subject":[],"published":{"date-parts":[[2021,7,7]]},"assertion":[{"value":"30 December 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"23 June 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"7 July 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Clinical data of the case was approved by the Institution Ethics Committee of PUMCH in accordance with the Declaration of Helsinki.The patient provided us written consent before hospitalization.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"145"}}