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dc.contributor.authorMamsen, Linn Salto
dc.contributor.authorKelsey, Thomas William
dc.contributor.authorErnst, Erik
dc.contributor.authorMacklon, Kirsten Tryde
dc.contributor.authorLund, Allan Meldgaard
dc.contributor.authorAndersen, Claus Yding
dc.date.accessioned2019-05-25T23:37:13Z
dc.date.available2019-05-25T23:37:13Z
dc.date.issued2018-07
dc.identifier.citationMamsen , L S , Kelsey , T W , Ernst , E , Macklon , K T , Lund , A M & Andersen , C Y 2018 , ' Cryopreservation of ovarian tissue may be considered in young girls with galactosemia ' , Journal of Assisted Reproduction and Genetics , vol. 35 , no. 7 , pp. 1209-1217 . https://doi.org/10.1007/s10815-018-1209-2en
dc.identifier.issn1058-0468
dc.identifier.otherPURE: 246459413
dc.identifier.otherPURE UUID: fd81a83e-9316-47b2-a1c6-28ba292e35f4
dc.identifier.otherPubMed: 29804175
dc.identifier.otherScopus: 85047409684
dc.identifier.otherORCID: /0000-0002-8091-1458/work/45366205
dc.identifier.otherPubMedCentral: PMC6063818
dc.identifier.otherWOS: 000440113200011
dc.identifier.urihttps://hdl.handle.net/10023/17761
dc.descriptionThis study was founded by Rigshospitalets Forskingspuljer, the Novo Nordisk Foundation, and ReproUnion.en
dc.description.abstractPurpose. The aim was to describe the first experience with fertility preservation by cryopreservation of ovarian tissue (OTC) in pre-pubertal girls with galactosemia and further to characterize ovarian follicular morphology and expression of proteins important for ovarian function. Methods. Retrospectively, follicle density was estimated in ovarian cortical tissues from 6 pre-pubertal girls below the age of 12 years diagnosed with galactosemia and from 31 girls below the age of 18 years who had one ovary removed for fertility preservation for other reasons prior to gonadotoxic treatment. Additionally, expression of 4 glycoproteins important for follicle development were analyzed with immunohistochemistry in two galactosemic ovaries (aged 0.9 and 1.7 years) and compared to normal age-matched controls. The proteins included were: anti-Müllerian hormone (AMH) pro-mature and C-terminal, growth differentiation factor-9 (GDF-9), bone morphogenetic protein 15 (BMP-15), and pregnancy-associated plasma protein A (PAPP-A). Results. Girls with galactosemia below the age of 5 years presented with morphological normal follicles and follicle densities within the 95% confidence interval (CI) of controls. No follicles were detected in the ovary from an 11.7-year-old girl with galactosemia. Expression of AMH, GDF-9, BMP-15, and PAPP-A appeared similar in follicles from girls with galactosemia and controls. Conclusions. These findings suggest that young girls with galactosemia maintain follicles in early childhood and fertility cryopreservation may be considered an option in this patient group. The pathophysiology of galactosemia leading to an accelerated follicle loss is unknown and it is currently unknown to what extent transplanted ovarian tissue can sustain fertility in adult life.
dc.format.extent9
dc.language.isoeng
dc.relation.ispartofJournal of Assisted Reproduction and Geneticsen
dc.rights© 2018, Springer Science+Business Media, LLC, part of Springer Nature. This work has been made available online in accordance with the publisher’s policies. This is the author created, accepted version manuscript following peer review and may differ slightly from the final published version. The final published version of this work is available at https://doi.org/10.1007/s10815-018-1209-2en
dc.subjectGalactosemiaen
dc.subjectPremature ovarian failureen
dc.subjectFertility preservationen
dc.subjectOvarian functionen
dc.subjectAMHen
dc.subjectPAPP-Aen
dc.subjectGDF-9en
dc.subjectQA75 Electronic computers. Computer scienceen
dc.subjectQH426 Geneticsen
dc.subjectRG Gynecology and obstetricsen
dc.subjectNDASen
dc.subject.lccQA75en
dc.subject.lccQH426en
dc.subject.lccRGen
dc.titleCryopreservation of ovarian tissue may be considered in young girls with galactosemiaen
dc.typeJournal articleen
dc.description.versionPostprinten
dc.contributor.institutionUniversity of St Andrews. School of Computer Scienceen
dc.contributor.institutionUniversity of St Andrews. Centre for Interdisciplinary Research in Computational Algebraen
dc.identifier.doihttps://doi.org/10.1007/s10815-018-1209-2
dc.description.statusPeer revieweden
dc.date.embargoedUntil2019-05-26


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