{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:12Z","timestamp":1784557032275,"version":"3.55.0"},"reference-count":16,"publisher":"Bioscientifica","issue":"1","license":[{"start":{"date-parts":[[2024,1,1]],"date-time":"2024-01-01T00:00:00Z","timestamp":1704067200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["edm.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2024,1,1]]},"abstract":"<jats:sec>\n                    <jats:title>Summary<\/jats:title>\n                    <jats:p>\n                      Functioning gonadotroph tumors are rare neoplasms that can cause ovarian hyperstimulation syndrome (OHSS) in women of reproductive age. Here, we present a case of a follicle-stimulating hormone (FSH)-producing pituitary neuroendocrine tumor (PitNET) with irregular menstrual cycles and OHSS in a Japanese woman. A 34-year-old woman with bilateral multi-cystic ovarian mass was referred to our hospital for ovarian surgery. The imaging feature of magnetic resonance imaging (MRI) of the ovary and elevated estradiol levels with normal FSH and low luteinizing hormone (LH) levels led us to suspect the presence of a functioning gonadotroph PitNET. MRI revealed a 19-mm pituitary tumor, and increased tracer uptake was observed in the pituitary lesion on\n                      <jats:sup>111<\/jats:sup>\n                      In-pentetreotide scintigraphy. Transsphenoidal tumor resection resulted in the resolution of the ovarian enlargement, normalization of her menstrual cycles, and spontaneous pregnancy. Immunohistochemistry (IHC) of the resected tumor for pituitary transcription factors, including steroidogenesis factor 1 (SF1) and estrogen receptor alpha, demonstrated positive immunoreactivity, whereas IHC for pituitary-specific positive transcription factor 1 was negative, suggesting that the tumor belonged to the SF1 lineage of PitNETs (gonadotroph tumor). The tumor cells showed positive expression of FSH\u03b2, while LH\u03b2 was mostly negative. Consistent with the high pituitary tumor uptake observed on\n                      <jats:sup>111<\/jats:sup>\n                      In-pentetreotide scintigraphy, the pituitary tumor showed positive expression of somatostatin receptor 2A. Detailed clinical and histological evaluations will provide useful information to understand these rare functioning gonadotroph tumors better.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Learning points<\/jats:title>\n                    <jats:list list-type=\"bullet\">\n                      <jats:list-item>\n                        <jats:p>Functioning gonadotroph tumors are very rare neuroendocrine tumors of pituitary origin.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Women of reproductive age presenting with bilateral multi-cystic ovarian enlargement, irregular menstrual cycles, and hyperestrogenemia under unsuppressed follicle-stimulating hormone (FSH) levels should be evaluated for FSH-producing tumor.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Raising awareness of OHSS due to functioning gonadotroph tumors is crucial to prevent unnecessary ovarian surgery.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Comprehensive histological analysis may provide useful information to better understand the characteristics of functioning gonadotroph tumors.<\/jats:p>\n                      <\/jats:list-item>\n                    <\/jats:list>\n                  <\/jats:sec>","DOI":"10.1530\/edm-23-0119","type":"journal-article","created":{"date-parts":[[2024,2,28]],"date-time":"2024-02-28T07:35:03Z","timestamp":1709105703000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-2","source":"Crossref","is-referenced-by-count":0,"title":["FSH-producing pituitary neuroendocrine tumor as a cause of ovarian hyperstimulation syndrome"],"prefix":"10.1530","volume":"2024","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1986-4808","authenticated-orcid":true,"given":"Takuya","family":"Kitamura","sequence":"first","affiliation":[{"name":"Department of Endocrinology and Metabolism, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"},{"name":"Department of Endocrinology, Metabolism, and Hypertension Research, Clinical Research Institute, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6475-5141","authenticated-orcid":true,"given":"Kazutaka","family":"Nanba","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"},{"name":"Department of Endocrinology, Metabolism, and Hypertension Research, Clinical Research Institute, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kento","family":"Doi","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naoya","family":"Kishimoto","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaoru","family":"Abiko","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryo","family":"Kuwahara","sequence":"additional","affiliation":[{"name":"Department of Radiology, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Koki","family":"Moriyoshi","sequence":"additional","affiliation":[{"name":"Department of Diagnostic Pathology, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naoko","family":"Inoshita","sequence":"additional","affiliation":[{"name":"Department of Pathology, Moriyama Memorial Hospital, Tokyo, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tetsuya","family":"Tagami","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"},{"name":"Department of Endocrinology, Metabolism, and Hypertension Research, Clinical Research Institute, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"bib1","series-title":"Cancers","first-page":"4324","article-title":"An overview of pituitary incidentalomas: diagnosis, clinical features, and management","volume":"14","author":"Tahara","year":"2022","unstructured":"Tahara SHattori YSuzuki KIshisaka ETeramoto S & Morita A. An overview of pituitary incidentalomas: diagnosis, clinical features, and management. Cancers2022144324. (https:\/\/doi.org\/10.3390\/cancers14174324)"},{"key":"bib2","series-title":"Endocrine Pathology","first-page":"6","article-title":"Overview of the 2022 WHO classification of pituitary tumors","volume":"33","author":"Asa","year":"2022","unstructured":"Asa SLMete OPerry A & Osamura RY. Overview of the 2022 WHO classification of pituitary tumors. Endocrine Pathology2022336\u201326. (https:\/\/doi.org\/10.1007\/s12022-022-09703-7)"},{"key":"bib3","series-title":"Medicina","first-page":"1071","article-title":"Updating the landscape for functioning gonadotroph tumors","volume":"58","author":"Ntali","year":"2022","unstructured":"Ntali G & Capatina C. Updating the landscape for functioning gonadotroph tumors. Medicina2022581071. (https:\/\/doi.org\/10.3390\/medicina58081071)"},{"key":"bib4","series-title":"World Neurosurgery","first-page":"e162","article-title":"Gonadotroph pituitary adenoma causing treatable infertility and ovarian hyperstimulation syndrome in female patients: neurosurgical, endocrinologic, gynecologic, and reproductive outcomes","volume":"150","author":"Hasegawa","year":"2021","unstructured":"Hasegawa HNesvick CLErickson DCohen SCYolcu YUKhan ZRaghunathan ANippoldt TBAtkinson JL & Van Gompel JJ. Gonadotroph pituitary adenoma causing treatable infertility and ovarian hyperstimulation syndrome in female patients: neurosurgical, endocrinologic, gynecologic, and reproductive outcomes. World Neurosurgery2021150e162\u2013e175. (https:\/\/doi.org\/10.1016\/j.wneu.2021.02.115)"},{"key":"bib5","series-title":"Pituitary","first-page":"454","article-title":"Functioning gonadotroph adenomas in premenopausal women: clinical and molecular characterization and review of the literature","volume":"25","author":"Wang","year":"2022","unstructured":"Wang LLiang HDeng CYu QGong FFeng FYou HLiang ZChen BDeng K, et al.Functioning gonadotroph adenomas in premenopausal women: clinical and molecular characterization and review of the literature. Pituitary202225454\u2013467. (https:\/\/doi.org\/10.1007\/s11102-021-01205-9)"},{"key":"bib6","series-title":"Clinical Endocrinology","first-page":"386","article-title":"Clinical characteristics of functioning gonadotroph adenoma in women presenting with ovarian hyperstimulation: audit of UK pituitary centres","volume":"99","author":"Papanikolaou","year":"2023","unstructured":"Papanikolaou NMillar OCoulden AParker NSit LKelly CCox JDhillo WSMeeran KAl Memar M, et al.Clinical characteristics of functioning gonadotroph adenoma in women presenting with ovarian hyperstimulation: audit of UK pituitary centres. Clinical Endocrinology202399386\u2013395. (https:\/\/doi.org\/10.1111\/cen.14949)"},{"key":"bib7","series-title":"JCEM Case Reports","first-page":"luad087","article-title":"Ovarian hyperstimulation syndrome caused by functional gonadotroph pituitary adenoma","volume":"1","author":"Tsukaguchi","year":"2023","unstructured":"Tsukaguchi RHasebe MHonjo S & Hamasaki A. Ovarian hyperstimulation syndrome caused by functional gonadotroph pituitary adenoma. JCEM Case Reports20231luad087. (https:\/\/doi.org\/10.1210\/jcemcr\/luad087)"},{"key":"bib8","series-title":"World Journal of Clinical Cases","first-page":"1341","article-title":"Functioning gonadotroph adenoma with hyperestrogenemia and ovarian hyperstimulation in a reproductive-aged woman: a case report and review of literature","volume":"11","author":"He","year":"2023","unstructured":"He YGao YT & Sun L. Functioning gonadotroph adenoma with hyperestrogenemia and ovarian hyperstimulation in a reproductive-aged woman: a case report and review of literature. World Journal of Clinical Cases2023111341\u20131348. (https:\/\/doi.org\/10.12998\/wjcc.v11.i6.1341)"},{"key":"bib9","series-title":"Cureus","first-page":"e26242","article-title":"A case of undiagnosed functional gonadotroph adenoma leading to ovarian hyperstimulation syndrome","volume":"14","author":"Mayur","year":"2022","unstructured":"Mayur OElshimy GBansal RJacob A & Raj R. A case of undiagnosed functional gonadotroph adenoma leading to ovarian hyperstimulation syndrome. Cureus202214e26242. (https:\/\/doi.org\/10.7759\/cureus.26242)"},{"key":"bib10","series-title":"International Journal of Women\u2019s Health","first-page":"427","article-title":"Continuous follicle-stimulating hormone exposure from pituitary adenoma causes periodic follicle recruitment and atresia, which mimics ovarian hyperstimulation syndrome","volume":"4","author":"Kanaya","year":"2012","unstructured":"Kanaya MBaba TKitajima YIkeda KShimizu AMorishita MHonnma HEndo T & Saito T. Continuous follicle-stimulating hormone exposure from pituitary adenoma causes periodic follicle recruitment and atresia, which mimics ovarian hyperstimulation syndrome. International Journal of Women\u2019s Health20124427\u2013431. (https:\/\/doi.org\/10.2147\/IJWH.S33386)"},{"key":"bib11","series-title":"Endotext","article-title":"Hyperprolactinemia","author":"Glezer","year":"2022","unstructured":"Glezer A & Bronstein MD. Hyperprolactinemia. In Endotext [Internet]. South Dartmouth,MA: MDText.com, Inc2022."},{"key":"bib12","series-title":"Internal Medicine","first-page":"545","article-title":"Follicle-stimulating hormone-secreting pituitary adenoma accompanied by painful thyroiditis","volume":"59","author":"Ichijo","year":"2020","unstructured":"Ichijo MTsuchiya KKasai TInoshita NYoshimoto HYamada S & Kitamura K. Follicle-stimulating hormone-secreting pituitary adenoma accompanied by painful thyroiditis. Internal Medicine202059545\u2013550. (https:\/\/doi.org\/10.2169\/internalmedicine.3667-19)"},{"key":"bib13","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"2407","article-title":"A gonadotroph adenoma with a high proportion of basic FSH isohormones by chromatofocusing","volume":"81","author":"Pigny","year":"1996","unstructured":"Pigny PHenric BLahlou NChristin SMazzuca MDewailly D & Boersma A. A gonadotroph adenoma with a high proportion of basic FSH isohormones by chromatofocusing. Journal of Clinical Endocrinology and Metabolism1996812407\u20132408. (https:\/\/doi.org\/10.1210\/jcem.81.6.8964889)"},{"key":"bib14","series-title":"Frontiers in Endocrinology","first-page":"626534","article-title":"Can inhibin B reflect ovarian reserve of healthy reproductive age women effectively?","volume":"12","author":"Wen","year":"2021","unstructured":"Wen JHuang KDu XZhang HDing TZhang CMa WZhong YQu WLiu Y, et al.Can inhibin B reflect ovarian reserve of healthy reproductive age women effectively?Frontiers in Endocrinology202112626534. (https:\/\/doi.org\/10.3389\/fendo.2021.626534)"},{"key":"bib15","series-title":"Pituitary","first-page":"553","article-title":"Ovarian hyperstimulation syndrome due to follicle-stimulating hormone-secreting pituitary adenomas","volume":"20","author":"Caretto","year":"2017","unstructured":"Caretto ALanzi RPiani CMolgora MMortini P & Losa M. Ovarian hyperstimulation syndrome due to follicle-stimulating hormone-secreting pituitary adenomas. Pituitary201720553\u2013560. (https:\/\/doi.org\/10.1007\/s11102-017-0817-7)"},{"key":"bib16","series-title":"StatPearls","article-title":"Octreotide scan","author":"Kapoor","year":"2021","unstructured":"Kapoor M & Kasi A. Octreotide scan. In StatPearls [Internet]. Treasure Island, FL: StatPearls Publishing2021."}],"container-title":["Endocrinology, Diabetes &amp; Metabolism Case Reports"],"original-title":[],"link":[{"URL":"https:\/\/edm.bioscientifica.com\/view\/journals\/edm\/2024\/1\/EDM23-0119.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/edm.bioscientifica.com\/downloadpdf\/journals\/edm\/2024\/1\/EDM23-0119.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/edm.bioscientifica.com\/downloadpdf\/journals\/edm\/2024\/1\/EDM23-0119.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T13:48:19Z","timestamp":1784555299000},"score":1,"resource":{"primary":{"URL":"http:\/\/journals.bioscientifica.com\/edm\/article\/3700"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,1,1]]},"references-count":16,"journal-issue":{"issue":"1"},"URL":"https:\/\/doi.org\/10.1530\/edm-23-0119","relation":{},"ISSN":["2052-0573"],"issn-type":[{"value":"2052-0573","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,1,1]]}}}