ARTICLE| Volume 46, ISSUE 6, P1017-1025, June 2023

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Deciding on future fertility: considerations of girls with Turner syndrome and their parents to opt for or against ovarian tissue cryopreservation

Open AccessPublished:March 03, 2023DOI:


      Research question

      What are the considerations of girls with Turner syndrome and their parents to opt for or against ovarian tissue cryopreservation (OTC)?


      Semi-structured in-depth interviews were conducted with girls with Turner syndrome and their parents until data saturation was reached. Participants were recruited through purposive sampling. Data were analysed using a thematic analysis approach.


      Thirteen parents and five girls who opted for OTC, and seven parents and three girls who declined OTC, were interviewed. Parents and girls mentioned that OTC offered hope, an opportunity to have genetic offspring and clarity about their current fertility status. Most participants were not afraid of the risks of surgery and trusted healthcare providers with this procedure. In contrast, families had to deal with uncertainties, owing to the lack of information on the success rate and long-term consequences of OTC in this group. Families indicated that they had to go through an important decision-making process in a short period of time, because of the limited number of participants in the OTC study.


      A new opportunity and hope for future fertility were considerations for opting for OTC. However, OTC also came with uncertainties owing to the experimental nature of this procedure in girls with Turner syndrome. Healthcare providers could share these experiences with girls with Turner syndrome and their parents to improve fertility-preservation counselling in this group.


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        • Bernard V.
        • Donadille B.
        • Zenaty D.
        • Courtillot C.
        • Salenave S.
        • Brac de la Perrière A.
        • Albarel F.
        • Fèvre A.
        • Kerlan V.
        • Brue T.
        • Delemer B.
        • Borson-Chazot F.
        • Carel J.C.
        • Chanson P.
        • Léger J.
        • Touraine P.
        • Christin-Maitre S.
        Spontaneous fertility and pregnancy outcomes amongst 480 women with Turner syndrome.
        Hum. Reprod. 2016; 31: 782-788
        • Borgström B.
        • Hreinsson J.
        • Rasmussen C.
        • Sheikhi M.
        • Fried G.
        • Keros V.
        • Fridström M.
        • Hovatta O.
        Fertility preservation in girls with turner syndrome: prognostic signs of the presence of ovarian follicles.
        J. Clin. Endocrinol. Metab. 2009; 94: 74-80
        • Braun V.
        • Clarke V.
        Using thematic analysis in psychology.
        Qualitative Research in Psychology. 2006; 3: 77-101
        • Dittrich R.
        • Hackl J.
        • Lotz L.
        • Hoffmann I.
        • Beckmann M.W.
        Pregnancies and live births after 20 transplantations of cryopreserved ovarian tissue in a single center.
        Fertil. Steril. 2015; 103: 462-468
        • Dolmans M.M.
        • Manavella D.D.
        Recent advances in fertility preservation.
        J. Obstet. Gynaecol. Res. 2019; 45: 266-279
        • Dolmans M.M.
        • von Wolff M.
        • Poirot C.
        • Diaz-Garcia C.
        • Cacciottola L.
        • Boissel N.
        • Liebenthron J.
        • Pellicer A.
        • Donnez J.
        • Andersen C.Y.
        Transplantation of cryopreserved ovarian tissue in a series of 285 women: a review of five leading European centers.
        Fertil. Steril. 2021; 115: 1102-1115
        • Folsom L.J.
        • Fuqua J.S.
        Reproductive Issues in Women with Turner Syndrome.
        Endocrinol Metab. Clin. North. Am. 2015; 44: 723-737
        • Galvin K.M.
        • Clayman M.L.
        Whose future is it? Ethical family decision making about daughters' treatment in the oncofertility context.
        Cancer Treat. Res. 2010; 156: 429-445
        • Ginsberg J.P.
        • Li Y.
        • Carlson C.A.
        • Gracia C.R.
        • Hobbie W.L.
        • Miller V.A.
        • Mulhall J.
        • Shnorhavorian M.
        • Brinster R.L.
        • Kolon T.F.
        Testicular tissue cryopreservation in prepubertal male children: an analysis of parental decision-making.
        Pediatr Blood Cancer. 2014; 61: 1673-1678
        • Gravholt C.H.
        • Andersen N.H.
        • Conway G.S.
        • Dekkers O.M.
        • Geffner M.E.
        • Klein K.O.
        • Lin A.E.
        • Mauras N.
        • Quigley C.A.
        • Rubin K.
        • Sandberg D.E.
        • Sas T.C.J.
        • Silberbach M.
        • Söderström-Anttila V.
        • Stochholm K.
        • van Alfen-van derVelden J.A.
        • Woelfle J.
        • Backeljauw P.F.
        Clinical practice guidelines for the care of girls and women with Turner syndrome: proceedings from the 2016 Cincinnati International Turner Syndrome Meeting.
        Eur. J. Endocrinol. 2017; 177: G1-G70
        • Grynberg M.
        • Bidet M.
        • Benard J.
        • Poulain M.
        • Sonigo C.
        • Cédrin-Durnerin I.
        • Polak M.
        Fertility preservation in Turner syndrome.
        Fertil. Steril. 2016; 105: 13-19
        • Hadnott T.N.
        • Gould H.N.
        • Gharib A.M.
        • Bondy C.A.
        Outcomes of spontaneous and assisted pregnancies in Turner syndrome: the U.S. National Institutes of Health experience.
        Fertil. Steril. 2011; 95: 2251-2256
        • Hershberger P.E.
        • Sipsma H.
        • Finnegan L.
        • Hirshfeld-Cytron J.
        Reasons Why Young Women Accept or Decline Fertility Preservation After Cancer Diagnosis.
        J. Obstet. Gynecol. Neonatal. Nurs. 2016; 45: 123-134
        • Hreinsson J.G.
        • Otala M.
        • Fridström M.
        • Borgström B.
        • Rasmussen C.
        • Lundqvist M.
        • Tuuri T.
        • Simberg N.
        • Mikkola M.
        • Dunkel L.
        • Hovatta O.
        Follicles are found in the ovaries of adolescent girls with Turner's syndrome.
        J. Clin. Endocrinol Metab. 2002; 87: 3618-3623
        • Jadoul P.
        • Guilmain A.
        • Squifflet J.
        • Luyckx M.
        • Votino R.
        • Wyns C.
        • Dolmans M.M.
        Efficacy of ovarian tissue cryopreservation for fertility preservation: lessons learned from 545 cases.
        Hum. Reprod. 2017; 32: 1046-1054
        • Li N.
        • Jayasinghe Y.
        • Kemertzis M.A.
        • Moore P.
        • Peate M.
        Fertility Preservation in Pediatric and Adolescent Oncology Patients: The Decision-Making Process of Parents.
        J. Adolesc Young Adult Oncol. 2017; 6: 213-222
        • McDougall R.J.
        • Gillam L.
        • Delany C.
        • Jayasinghe Y.
        Ethics of fertility preservation for prepubertal children: should clinicians offer procedures where efficacy is largely unproven?.
        J. Med. Ethics. 2018; 44: 27-31
        • Modi D.N.
        • Sane S.
        • Bhartiya D.
        Accelerated germ cell apoptosis in sex chromosome aneuploid fetal human gonads.
        Mol. Hum. Reprod. 2003; 9: 219-225
        • Oktay K.
        • Bedoschi G.
        • Berkowitz K.
        • Bronson R.
        • Kashani B.
        • McGovern P.
        • Pal L.
        • Quinn G.
        • Rubin K.
        Fertility Preservation in Women with Turner Syndrome: A Comprehensive Review and Practical Guidelines.
        J. Pediatr. Adolesc. Gynecol. 2016; 29: 409-416
        • Pasquino A.M.
        • Passeri F.
        • Pucarelli I.
        • Segni M.
        • Municchi G.
        Spontaneous pubertal development in Turner's syndrome. Italian Study Group for Turner's Syndrome.
        J. Clin. Endocrinol. Metab. 1997; 82: 1810-1813
        • Peek R.
        • Schleedoorn M.
        • Smeets D.
        • van de Zande G.
        • Groenman F.
        • Braat D.
        • van der Velden J.
        • Fleischer K.
        Ovarian follicles of young patients with Turner's syndrome contain normal oocytes but monosomic 45,X granulosa cells.
        Hum. Reprod. 2019; 34: 1686-1696
        • Reynaud K.
        • Cortvrindt R.
        • Verlinde F.
        • De Schepper J.
        • Bourgain C.
        • Smitz J.
        Number of ovarian follicles in human fetuses with the 45,X karyotype.
        Fertil. Steril. 2004; 81: 1112-1119
        • Saenger P.
        Turner's syndrome.
        N. Engl. J. Med. 1996; 335: 1749-1754
        • Schleedoorn M.J.
        • Mulder B.H.
        • Braat D.D.M.
        • Beerendonk C.C.M.
        • Peek R.
        • Nelen W.
        • Van Leeuwen E.
        • Van der Velden A.
        • Fleischer K.
        • Turner Fertility Expert Panel O.
        International consensus: ovarian tissue cryopreservation in young Turner syndrome patients: outcomes of an ethical Delphi study including 55 experts from 16 different countries.
        Hum. Reprod. 2020; 35: 1061-1072
        • Schleedoorn M.J.
        • van der Velden A.A.E.M.
        • Braat D.D.M.
        • Peek R.
        • Fleischer K.
        To Freeze or Not to Freeze? An Update on Fertility Preservation In Females with Turner Syndrome.
        Pediatr. Endocrinol Rev. 2019; 16: 369-382
        • Schleedoorn M.J.
        • van der Velden J.
        • Braat D.
        • Beerendonk I.
        • van Golde R.
        • Peek R.
        • Fleischer K.
        TurnerFertility trial: PROTOCOL for an observational cohort study to describe the efficacy of ovarian tissue cryopreservation for fertility preservation in females with Turner syndrome.
        BMJ. Open. 2019; 9e030855
        • Stochholm K.
        • Juul S.
        • Juel K.
        • Naeraa R.W.
        • Gravholt C.H.
        Prevalence, incidence, diagnostic delay, and mortality in Turner syndrome.
        J. Clin. Endocrinol. Metab. 2006; 91: 3897-3902
        • Sullivan-Pyke C.S.
        • Carlson C.A.
        • Prewitt M.
        • Gracia C.R.
        • Ginsberg J.P.
        Ovarian tissue cryopreservation (OTC) in prepubertal girls and young women: an analysis of parents' and patients' decision-making.
        J. Assist. Reprod. Genet. 2018; 35: 593-600
        • Sutton E.J.
        • McInerney-Leo A.
        • Bondy C.A.
        • Gollust S.E.
        • King D.
        • Biesecker B.
        Turner syndrome: four challenges across the lifespan.
        Am. J. Med. Genet. A. 2005; 139a: 57-66
        • Sylvén L.
        • Magnusson C.
        • Hagenfeldt K.
        • von Schoultz B.
        Life with Turner's syndrome–a psychosocial report from 22 middle-aged women.
        Acta Endocrinol (Copenh). 1993; 129: 188-194
        • Tanaka T.
        • Igarashi Y.
        • Ozono K.
        • Ohyama K.
        • Ogawa M.
        • Osada H.
        • Onigata K.
        • Kanzaki S.
        • Kohno H.
        • Seino Y.
        • Takahashi H.
        • Tajima T.
        • Tachibana K.
        • Tanaka H.
        • Nishi Y.
        • Hasegawa T.
        • Fujita K.
        • Yorifuji T.
        • Horikawa R.
        • Yokoya S.
        Frequencies of spontaneous breast development and spontaneous menarche in Turner syndrome in Japan.
        Clin. Pediatr. Endocrinol. 2015; 24: 167-173
        • Tong A.
        • Sainsbury P.
        • Craig J.
        Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups.
        Int. J. Qual. Health Care. 2007; 19: 349-357
        • van der Coelen S.
        • Schleedoorn M.J.
        • Nadesapillai S.
        • Peek R.
        • Braat D.D.M.
        • van der Velden A.A.E.M.
        • Fleischer K.
        • Oerlemans A.J.M.
        Evaluation of the decision-making process on ovarian tissue cryopreservation in girls with Turner syndrome by patients, parents and caregivers.
        Human Reproduction. 2022;
        • Van der Ven H.
        • Liebenthron J.
        • Beckmann M.
        • Toth B.
        • Korell M.
        • Krüssel J.
        • Frambach T.
        • Kupka M.
        • Hohl M.K.
        • Winkler-Crepaz K.
        • Seitz S.
        • Dogan A.
        • Griesinger G.
        • Häberlin F.
        • Henes M.
        • Schwab R.
        • Sütterlin M.
        • von Wolff M.
        • Dittrich R.
        Ninety-five orthotopic transplantations in 74 women of ovarian tissue after cytotoxic treatment in a fertility preservation network: tissue activity, pregnancy and delivery rates.
        Hum. Reprod. 2016; 31: 2031-2041


      Sapthami Nadesapillai is a physician in reproductive medicine at the Department of Obstetrics and Gynaecology of the Radboudumc, the Netherlands. She is currently combining her clinical work with a PhD project that focuses on different aspects of fertility preservation in girls with Turner syndrome.
      Key message
      Making a decision about ovarian tissue cryopreservation is challenging for girls with Turner syndrome and their parents. This experimental procedure offered hope for genetic offspring, but outcome and long-term consequences remain uncertain. Physicians can use our findings to improve fertility preservation counselling in girls with Turner syndrome.