The decision-making process regarding ovarian tissue cryopreservation in girls with Turner syndrome by patients, parents, and healthcare providers: a mixed-methods study
Open accessAbstract
INTRODUCTION: Ovarian tissue cryopreservation (OTC) has proven to be effective in other patient groups, but the effectiveness in girls with Turner syndrome (TS) is still unclear. Guidelines for counselling about OTC in TS are lacking. The aim of this study was to gain insight into the experiences of patients, parents, and healthcare providers with the decision-making process regarding OTC in girls with TS. METHODS: Within a year after counselling, a survey was sent to 132 girls with TS and their parents. Furthermore, focus groups were conducted with (1) gynaecologists with subspeciality reproductive medicine, (2) paediatric endocrinologists, (3) parents of girls aged 2-12, and (4) parents of girls aged 13-18. Transcripts were analysed using a thematic analysis approach. RESULTS: The response rate of the survey was 45%. Of the survey respondents, 90% appreciated counselling regarding their future parenting options and considered it an addition to existing healthcare. Girls with TS and their parents indicated that the option of OTC raised hope for future genetic offspring and instantly made them feel that their only option was to seize this opportunity. Gynaecologists and paediatricians found it challenging to truly make families grasp a realistic perspective of OTC in girls with TS. DISCUSSION AND CONCLUSION: Offering young girls with TS the possibility of fertility preservation in an experimental setting raised high hopes and led to challenges for healthcare providers in ensuring a considered decision. The appropriate moment for counselling should be tailored to the individual and discussed with patient, parents, and paediatrician.
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
Individuals with TS may derive comfort from learning about fertility preservation options through consultation with knowledgeable providers and the associated sense of having explored all possible options for future fertility.
“Individuals with TS may derive comfort from learning about fertility preservation options through consultation with knowledgeable providers and the associated sense of having explored all possible options for future fertility.572,622”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [572]
Early referral for fertility preservation, including for pre-adolescent or adolescent individuals with TS, might improve outcomes.
“Early referral for fertility preservation—including for pre-adolescent or adolescent individuals with TS—might improve outcomes572,623 and allow time to consider goals/implications and the possibility of fertility preservation.”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [572]
There are unique barriers to utilizing fertility preservation procedures for children and adolescents with TS, including the relative dearth of physicians with expertise in pediatric fertility care, the difficulty of assessing future family building goals for younger patients, and concerns about pre-adolescent/adolescent patients' ability to tolerate interventions such as ovarian stimulation or oo
“For pediatric clinicians, reducing harm includes acknowledging unique barriers to utilizing fertility preservation procedures for children and adolescents. These include: the relative dearth of physicians with expertise in pediatric fertility care; the difficulty of assessing future family building goals for younger patients; and concerns about pre-adolescent/adolescent patients’ ability to”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [572]
Self-check
Percentages, sums and reported differences re-computed from the paper’s own numbers.
The self-check needs the paper’s full text.
Cites
Using thematic analysis in psychology
Braun, V., & Clarke, V. (2006) · Qualitative Research in Psychology · doi:10.1191/1478088706qp063oa · Cited by 194,414 · see citing works
Tong, A., Sainsbury, P., & Craig, J. (2007) · International Journal for Quality in Health Care · doi:10.1093/intqhc/mzm042 · Cited by 42,509 · see citing works
Qualitative research in health care: Assessing quality in qualitative research
Mays, N. (2000) · BMJ · doi:10.1136/bmj.320.7226.50 · Cited by 3,742 · see citing works
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., et al. (2017) · European Journal of Endocrinology · doi:10.1530/eje-17-0430 · Cited by 1,101 · see citing works
Fertility Preservation in Women
Donnez, J., & Dolmans, M. (2017) · New England Journal of Medicine · doi:10.1056/nejmra1614676 · Cited by 892 · see citing works
Prevalence, incidence, diagnostic delay, and mortality in Turner syndrome
Stochholm, K., Juul, S., Juel, K., Naeraa, R. W., & Højbjerg Gravholt, C. (2006) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2006-0558 · Cited by 652 · see citing works
Letourneau, J. M., Ebbel, E. E., Katz, P., et al. (2011) · Cancer · doi:10.1002/cncr.26459 · Cited by 565 · see citing works
Live birth after autograft of ovarian tissue cryopreserved during childhood: Figure 1
Demeestere, I., Simon, P., Dedeken, L., Moffa, F., Tsépélidis, S., Brachet, C., Delbaere, A., Devreker, F., & Ferster, A. (2015) · Human Reproduction · doi:10.1093/humrep/dev128 · Cited by 400 · see citing works
Turner's Syndrome in Adulthood
Elsheikh, M., Dunger, D. B., Conway, G. S., & Wass, J. A. H. (2002) · Endocrine Reviews · doi:10.1210/edrv.23.1.0457 · Cited by 397 · see citing works
Dolmans, M., von Wolff, M., Poirot, C., Díaz‐García, C., Cacciottola, L., Boissel, N., Liebenthron, J., Pellicer, A., Donnez, J., & Andersen, C. Y. (2021) · Fertility and Sterility · doi:10.1016/j.fertnstert.2021.03.008 · Cited by 341 · see citing works
Spontaneous fertility and pregnancy outcomes amongst 480 women with Turner syndrome
Bernard, V., Donadille, B., Zenaty, D., Courtillot, C., Salenave, S., Brac de la Perrière, A., Albarel, F., Fèvre, A., Kerlan, V., Brue, T., et al. (2016) · Human Reproduction · doi:10.1093/humrep/dew012 · Cited by 266 · see citing works
Birgit, B., Julius, H., Carsten, R., Maryam, S., Gabriel, F., Victoria, K., Margareta, F., & Outi, H. (2009) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jc.2008-0708 · Cited by 254 · see citing works
Prasath, E. B., Chan, M. L. H., Wong, W., et al. (2013) · Human Reproduction · doi:10.1093/humrep/det420 · Cited by 214 · see citing works
Oktay, K., Bedoschi, G., Berkowitz, K. M., BRONSON, R. A., Kashani, B. N., McGovern, P. G., Pal, L., Quinn, G. P., & Rubin, K. (2015) · Journal of Pediatric and Adolescent Gynecology · doi:10.1016/j.jpag.2015.10.011 · Cited by 210 · see citing works
Follicles Are Found in the Ovaries of Adolescent Girls with Turner's Syndrome
Hreinsson, J. G., Otala, M., Fridström, M., Borgström, B., Rasmussen, C., Lundqvist, M., Tuuri, T., Simberg, N. H., Mikkola, M., Dunkel, L., et al. (2002) · The Journal of Clinical Endocrinology & Metabolism · doi:10.1210/jcem.87.8.8753 · Cited by 192 · see citing works
Turner syndrome: four challenges across the lifespan
Sutton, E. J., McInerney‐Leo, A., Bondy, C. A., Gollust, S. E., King, D., & Biesecker, B. (2005) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.30911 · Cited by 171 · see citing works
Fertility preservation in Turner syndrome
Grynberg, M., Bidet, M., Bénard, J., Poulain, M., Sonigo, C., Cédrin‐Durnerin, I., & Polak, M. (2015) · Fertility and Sterility · doi:10.1016/j.fertnstert.2015.11.042 · Cited by 124 · see citing works
The Imperative Character of Medical Technology and the Meaning of “Anticipated Decision Regret”
Tymstra, T. (1989) · International Journal of Technology Assessment in Health Care · doi:10.1017/s0266462300006437 · Cited by 121 · see citing works
Wyns, C., Collienne, C., Shenfield, F., Robert, A. R., Laurent, P., Roegiers, L., & Brichard, B. (2015) · Human Reproduction · doi:10.1093/humrep/dev161 · Cited by 107 · see citing works
Young Adult Female Cancer Survivors' Decision Regret About Fertility Preservation
Benedict, C., Thom, B., & Kelvin, J. F. (2015) · Journal of Adolescent and Young Adult Oncology · doi:10.1089/jayao.2015.0002 · Cited by 82 · see citing works
Parental desire and acceptability of spermatogonial stem cell cryopreservation in boys with cancer
van den Berg, H., Repping, S., & van der Veen, F. (2006) · Human Reproduction · doi:10.1093/humrep/del375 · Cited by 75 · see citing works
Fertility, pregnancy, and medical management of Turner syndrome in the reproductive years
Karnis, M. F. (2012) · Fertility and Sterility · doi:10.1016/j.fertnstert.2012.08.022 · Cited by 68 · see citing works
Peek, R., Schleedoorn, M., Smeets, D., van de Zande, G., Groenman, F., Braat, D., van der Velden, J., & Fleischer, K. (2019) · Human Reproduction · doi:10.1093/humrep/dez135 · Cited by 67 · see citing works
Self-citationMcDougall, R. J., Gillam, L. H., Delany, C. M., et al. (2017) · Journal of Medical Ethics · doi:10.1136/medethics-2016-104042 · Cited by 61 · see citing works
Li, N., Jayasinghe, Y., Kemertzis, M. A., et al. (2016) · Journal of Adolescent and Young Adult Oncology · doi:10.1089/jayao.2016.0061 · Cited by 46 · see citing works
Schleedoorn, M. J., Mulder, B. H., Braat, D. D. M., Beerendonk, C. C. M., Peek, R., Nelen, W. L. D. M., Van Leeuwen, E., Van der Velden, A. A. E. M., & Fleischer, K. (2020) · Human Reproduction · doi:10.1093/humrep/deaa007 · Cited by 43 · see citing works
Self-citationNadesapillai, S., van der Velden, J., Smeets, D., van de Zande, G., Braat, D., Fleischer, K., & Peek, R. (2021) · Fertility and Sterility · doi:10.1016/j.fertnstert.2020.11.006 · Cited by 31 · see citing works
Self-citationVergier, J., Bottin, P., Saias, J., Reynaud, R., Guillemain, C., & Courbiere, B. (2019) · Clinical Endocrinology · doi:10.1111/cen.14076 · Cited by 30 · see citing works
Morgan, T. L., Kapa, H. M., Crerand, C. E., Kremen, J., Tishelman, A. C., Davis, S., & Nahata, L. (2019) · Fertility and Sterility · doi:10.1016/j.fertnstert.2019.05.010 · Cited by 26 · see citing works
Schleedoorn, M., van der Velden, J., Braat, D., Beerendonk, I., van Golde, R., Peek, R., & Fleischer, K. (2019) · BMJ Open · doi:10.1136/bmjopen-2019-030855 · Cited by 26 · see citing works
Self-citationTo Freeze or Not to Freeze? An Update on Fertility Preservation In Females with Turner Syndrome.
Schleedoorn, M., van der Velden, J. A. E. M., Braat, D. D. M., Peek, R., & Fleischer, K. (2019) · PubMed · doi:10.17458/per.vol16.2019.svb.tofreezeornot · Cited by 21 · see citing works
Self-citationNawroth, F., Schüring, A. N., & von Wolff, M. (2020) · Acta Obstetricia Et Gynecologica Scandinavica · doi:10.1111/aogs.13984 · Cited by 19 · see citing works
Reproductive medicine and Turner syndrome: ethical issues
Wasserman, D., & Asch, A. (2012) · Fertility and Sterility · doi:10.1016/j.fertnstert.2012.08.036 · Cited by 15 · see citing works
Sullivan-Pyke, C. S., Carlson, C. A., Prewitt, M., Gracia, C. R., & Ginsberg, J. P. (2018) · Journal of Assisted Reproduction and Genetics · doi:10.1007/s10815-018-1123-7 · Cited by 15 · see citing works
Karyotype is associated with timing of ovarian failure in women with Turner syndrome
Fitz, V. W., Law, J. R., & Peavey, M. (2021) · Journal of Pediatric Endocrinology and Metabolism · doi:10.1515/jpem-2020-0304 · Cited by 11 · see citing works
Hamama‐Raz, Y., Ginossar-David, E., & Ben‐Ezra, M. (2016) · Israel Journal of Health Policy Research · doi:10.1186/s13584-016-0116-1 · Cited by 7 · see citing works
Chan, S.-W., Cipres, D. T., Katz, A., et al. (2014) · Fertility and Sterility · doi:10.1016/j.fertnstert.2014.07.556 · Cited by 5 · see citing works
Flagged first, then by citations
Research works printed but not linked: 1
- [38]Mason J Qualitative researching. Chapter 4 Generating qualitative data: observation, documents and visual data 2002 Sage Publications Inc
Cited by
Sorted by citations
Clinical practice guidelines for the care of girls and women with Turner syndrome.
Gravholt, C. H., Andersen, N. H., Christin-Maitre, S., Davis, S. M., Duijnhouwer, A., Gawlik, A., Maciel-Guerra, A. T., Gutmark-Little, I., Fleischer, K., Hong, D., et al. (2024) · European Journal of Endocrinology · doi:10.1093/ejendo/lvae050 · Cited by 224 · see citing works
Self-citationRodriguez-Wallberg, K. A., Sergouniotis, F., Nilsson, H. P., & Lundberg, F. E. (2023) · Frontiers in Endocrinology · doi:10.3389/fendo.2023.1135249 · Cited by 29 · see citing works
Nadesapillai, S., Mol, F., Broer, S. L., Stevens Brentjens, L. B. P. M., Verhoeven, M. O., Heida, K. Y., Goddijn, M., van Golde, R. J. T., Bos, A. M. E., van der Coelen, S., et al. (2023) · Journal of Clinical Medicine · doi:10.3390/jcm12206502 · Cited by 13 · see citing works
Self-citation
- Authors
- van der Coelen, S., van der Velden, J., Nadesapillai, S., Peek, R., Braat, D., Schleedoorn, M., Fleischer, K., & Oerlemans, A.
- Affiliations
- Radboud University Nijmegen (NL)
- ORCID
- Venue
- Hormone Research in Paediatrics
- Publisher
- S. Karger AG
- Published
- 2022
- Volume
- 95
- Issue
- 4
- Pages
- 374-383
- ISSN
- 1663-2818 (ISSN-L)
- 1663-2826
- PMC
- PMC9677842
- OpenAlex
- W4281725825
- Cited by
- 22
- References
- 37
- Citations by year
- 2026: 4 · 2025: 6 · 2024: 5 · 2023: 6 · 2022: 1
- FWCI
- 3.97
- Type
- journal article
- Language
- en
- Open access
- hybrid
- doi.org
- Licence
- Locations
- Hormone Research in Paediatrics (publishedVersion, cc-by-nc)
- Research Square (acceptedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by-nc)
- PubMed (publishedVersion)
- Radboud Repository (Radboud University) (submittedVersion)
- Radboud Repository (Radboud University) (submittedVersion, other-oa)
- Topics
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Reproductive Biology and Fertility
- Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Ethics and Legal Issues in Pediatric Healthcare
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Clinical Aspects of Sex Determination and Chromosomal Abnormalities
- Keywords
- ovarian tissue cryopreservation, Turner syndrome, fertility preservation, decision-making, patient counseling, parents, healthcare workers, girls, reproductive medicine, pediatric endocrinology, hope and expectation
- MeSH terms
- Child, Female, Health Personnel, Humans, Parents, Turner Syndrome, Cryopreservation, Fertility Preservation
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review
