Recombinant Human FSH Treatment Outcomes in Five Boys With Severe Congenital Hypogonadotropic Hypogonadism

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dc.contributor University of Helsinki, Raivio Group en
dc.contributor University of Helsinki, Raivio Group en
dc.contributor University of Helsinki, Centre of Excellence in Stem Cell Metabolism en
dc.contributor University of Helsinki, Raivio Group en
dc.contributor University of Helsinki, Department of Physiology en
dc.contributor.author Kohva, Ella
dc.contributor.author Huopio, Hanna
dc.contributor.author Hero, Matti
dc.contributor.author Miettinen, Päivi J.
dc.contributor.author Vaaralahti, Kirsi
dc.contributor.author Sidoroff, Virpi
dc.contributor.author Toppari, Jorma
dc.contributor.author Raivio, Taneli
dc.date.accessioned 2019-01-15T12:03:01Z
dc.date.available 2019-01-15T12:03:01Z
dc.date.issued 2018-10-15
dc.identifier.citation Kohva , E , Huopio , H , Hero , M , Miettinen , P J , Vaaralahti , K , Sidoroff , V , Toppari , J & Raivio , T 2018 , ' Recombinant Human FSH Treatment Outcomes in Five Boys With Severe Congenital Hypogonadotropic Hypogonadism ' , Journal of the Endocrine Society , vol. 2 , no. 12 , pp. 1345-1356 . https://doi.org/10.1210/js.2018-00225 en
dc.identifier.issn 2472-1972
dc.identifier.other PURE: 120623495
dc.identifier.other PURE UUID: 5493e4eb-6a77-4721-b6e0-b6ed8ae5aa4e
dc.identifier.other ORCID: /0000-0002-5184-9616/work/52696882
dc.identifier.other WOS: 000455534800003
dc.identifier.uri http://hdl.handle.net/10138/292439
dc.description.abstract Context Recombinant human FSH (r-hFSH), given to prepubertal boys with hypogonadotropic hypogonadism (HH), may induce Sertoli cell proliferation and thereby increase sperm-producing capacity later in life. Objective To evaluate the effects of r-hFSH, human chorionic gonadotropin (hCG), and testosterone (T) in such patients. Design and Setting Retrospective review in three tertiary centers in Finland between 2006 and 2016. Patients Five boys: ANOS1 mutation in two, homozygous PROKR2 mutation in one, FGFR1 mutation in one, and homozygous GNRHR mutation in one. Prepubertal testicular volume (TV) varied between 0.3 and 2.3 mL; three boys had micropenis, three had undergone orchidopexy. Interventions Two boys received r-hFSH (6 to 7 months) followed by r-hFSH plus hCG (33 to 34 months); one received T (6 months), then r-hFSH plus T (29 months) followed by hCG (25 months); two received T (3 months) followed by r-hFSH (7 months) or r-hFSH plus T (8 months). Main Outcome Measures TV, inhibin B, anti-Müllerian hormone, T, puberty, sperm count. Results r-hFSH doubled TV (from a mean ± SD of 0.9 ± 0.9 mL to 1.9 ± 1.7 mL; P < 0.05) and increased serum inhibin B (from 15 ± 5 ng/L to 85 ± 40 ng/L; P < 0.05). hCG further increased TV (from 2.1 ± 2.3 mL to 8.6 ± 1.7 mL). Two boys with initially extremely small testis size (0.3 mL) developed sperm (maximal sperm count range, 2.8 to 13.8 million/mL), which was cryopreserved. Conclusions Spermatogenesis can be induced with gonadotropins even in boys with HH who have extremely small testes, and despite low-dose T treatment given in early puberty. Induction of puberty with gonadotropins allows preservation of fertility fi
dc.format.extent 12
dc.language.iso eng
dc.relation.ispartof Journal of the Endocrine Society
dc.rights en
dc.subject 3121 General medicine, internal medicine and other clinical medicine en
dc.title Recombinant Human FSH Treatment Outcomes in Five Boys With Severe Congenital Hypogonadotropic Hypogonadism en
dc.type Article
dc.description.version Peer reviewed
dc.identifier.doi https://doi.org/10.1210/js.2018-00225
dc.type.uri info:eu-repo/semantics/other
dc.type.uri info:eu-repo/semantics/publishedVersion
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