Characterization of follicles in girls and young women with Turner syndrome who underwent ovarian tissue cryopreservation
Open accessAbstract
Objective To characterize ovarian follicles of girls and young women with Turner syndrome (TS) who underwent ovarian tissue cryopreservation (OTC).Design Retrospective case-control study.Setting University hospital.Patient(s) Fifteen girls and young women with TS aged 5-22 years at OTC were included, together with 42 control girls and young women aged 1-25 years who underwent OTC because of cancer.Intervention(s) None.Main outcome measure(s) Follicle density (follicles/mm 3), morphology, and health were assessed in ovarian cortex biopsies from TS patients and compared with controls. Hormone concentrations were measured in serum and follicle fluids. Immature cumulus oocyte complexes were obtained and matured in vitro.Result(s) Follicles were found in 60% of the biopsies (9 of 15) from TS ovaries. In 78% of the ovaries (7 of 9) with follicles, the follicle density was within the 95% confidence interval of the control group. There was a high rate of abnormal follicle morphology. Six follicle-specific proteins were expressed similarly in TS and control ovaries. However, apoptosis and zona pellucida protein expression were found to be abnormal in TS. Turner syndrome follicle fluid from small antral follicles had lower concentrations of estrogen and testosterone and higher concentrations of antimüllerian hormone than controls. Thirty-one cumulus oocyte complexes were collected from one patient and cultured for 48 hours in vitro, resulting in five metaphase II oocytes (maturation rate 16%, degeneration rate 19%).Conclusion(s) The benefits of OTC may be limited to a highly selected group of TS mosaic patients in whom a sizeable pool of normal follicles is present at OTC.
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Findings associated with an increased likelihood of identifying follicles in the ovarian cortex include mosaic karyotype with a 46,XX cell line, spontaneous puberty, measurable AMH, or FSH <10 IU/L.
“Findings that may be associated with an increased likelihood of identifying follicles in the ovarian cortex include (1) mosaic karyotype with a 46,XX cell line, (2) spontaneous puberty, (3) measurable AMH or (4) FSH <10 IU L−1.263,578,579”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
In individuals with TS, AMH is associated with clinical features of ovarian reserve and has the strongest positive correlation with the presence of follicles in ovarian cortex tissue.
“In individuals with TS, AMH is associated with clinical features of ovarian reserve270,581 and has the strongest positive correlation with the presence of follicles in ovarian cortex tissue .263,579,582”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
Current knowledge on OTC in TS consists of four cohort studies and four case reports, involving a total of 185 TS patients with ages varying between 3 and 22 years.
“The current knowledge on OTC in TS consists of four cohort studies and four case reports, involving a total of 185 TS-patients with ages varying between 3 and 22 years.263,579,582,614”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
Individuals with mosaic TS are more likely to have spontaneous puberty, normal gonadotropin levels, measurable AMH, and follicles in ovarian biopsies compared to those with monosomy X karyotype.
“Individuals with mosaic TS are more likely to have spontaneous puberty, normal gonadotropin levels, a measurable AMH, and follicles in ovarian biopsies as compared to those who have monosomy X karyotype.263,579,608”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
Aberrant follicle morphology of up to 30%-67% was observed in TS ovaries.
“Additionally, an aberrant follicle morphology of up to 30%-67% was observed in TS ovaries.579,609”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
Mamsen et al. studied retrospectively histology sections of 15 TS patients who underwent OTC and in 9/15 patients (60%) follicles were found.
“Mamsen et al. studied retrospectively histology sections of 15 TS patients who underwent OTC and in 9/15 patients (60%) follicles were found.579”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
Follicles were present in 50%-100% of TS girls with FSH <10 IU/L (including prepubertal girls).
“Follicles were present in 50%-100% of TS girls with FSH <10 IU L−1 (including prepubertal girls).263,579,582”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
Spontaneous puberty has a significant positive correlation with the presence of follicles.
“Spontaneous puberty has a significant positive correlation with the presence of follicles.263,579,582,618”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
The odds of finding follicles in patients with a structural X chromosome aberration or a 45,X/47,XXX karyotype is 23%-44%, and with a 45,X karyotype 4%-11%.
“The odds of finding follicles in patients with a structural X chromosome aberration or a 45,X/47,XXX karyotype is 23%-44% and with a 45, X karyotype 4%-11%.263,579,582”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
Patients with a mosaic karyotype have the highest prevalence of follicles, 67%-100%.
“Patients with a mosaic karyotype have the highest prevalence of follicles, respectively 67%-100%.263,579,582”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
FSH <10 IU/L has a positive correlation with the presence of follicles.
“An FSH <10 IU L−1 has a positive correlation with the presence of follicles.”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [579]
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Research works printed but not linked: 1
- Berjonneau C, Rives A, Jolly-H elas G, Cuny A, Castanet M, Letailleur M, et al. Management of fertility preservation in young patients with Turner syn- drome. Vienna, Austria: International Society For Fertility Preservation (ISFP) Conference; 2017:1. on de folículos en ni ñas y mujeres j ovenes con síndrome de Turner que realizaron criopreservaci on de tejido ov arico Objetivo: Caracterizar folículos ov aricos de niñas y mujeres j ovenes con síndrome de Turner (ST) que realizaron criopreservaci on de tejido ov arico (CTO). Diseño: Estudio retrospectivo de casos y controles. Ambito: Hospital universitario. Paciente(s): Quince niñas y mujeres j ovenes con ST de 5-22 años en la CTO fueron incluidas, junto con 42 niñas y mujeres j ovenes controles de 1-25 años quienes realizaron CTO por c ancer. Intervenci on(es): Ninguna. Principal(es) medida(s) de resultado: Densidad folicular (folículos/mm 3 ), morfología, y estado fueron evaluados en las biopsias de corteza ov arica de pacientes con ST y comparadas con controles. Concentraciones hormonales fueron medidas en fluidos s erico y fo- licular. Complejos c umulo ovocito inmaduros fueron obtenidos y madurados in vitro. Resultado(s): Fueron encontrados folículos en 60% de las biopsias (9 de 15) ovarios con ST. En el 78% de los ovarios (7 de 9) con folículos, la densidad folicular estuvo dentro del intervalo de confianza del 95% del grupo control. Hubo una alta tasa de morfología folicular anormal. Seis proteínas foliculares específicas fueron expresadas de manera similar en ovarios con ST y controles. Sin em- bargo, apoptosis y la expresi on de proteínas de la zona pel ucida fueron halladas anormales en ST. El fluido folicular de folículos antrales pequeños en ST tuvo concentraciones m as bajas de estr ogeno y T y concentraciones m as altas de hormona anti-m€ ulleriana que los con- troles. Treinta y un complejos c umulo ovocito fueron recolectados de una paciente y cultivados durante 48 horas in vitro, resultando en cinco ovocitos metafase II (tasa de maduraci on 16%, tasa de degeneraci on 9%).
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- Authors
- Mamsen, L. S., Charkiewicz, K., Anderson, R. A., Telfer, E. E., McLaughlin, M., Kelsey, T. W., Kristensen, S. G., Gook, D. A., Ernst, E., & Andersen, C. Y.
- Affiliations
- University of Copenhagen (DK)
- MRC Centre for Reproductive Health (GB)
- University of Edinburgh (GB)
- Royal Women's Hospital (AU)
- Regional Hospital Randers (DK)
- Corresponding author
- Mamsen, Linn Salto
- ORCID
- Venue
- Fertility and Sterility
- Publisher
- Elsevier BV
- Published
- 2019-06
- Volume
- 111
- Issue
- 6
- Pages
- 1217-1225.e3
- ISSN
- 0015-0282 (ISSN-L)
- OpenAlex
- W2923727527
- Cited by
- 86
- References
- 33
- Citations by year
- 2026: 8 · 2025: 9 · 2024: 10 · 2023: 16 · 2022: 15 · 2021: 10 · 2020: 14 · 2019: 4
- FWCI
- 9.91
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- Locations
- Fertility and Sterility (publishedVersion)
- PubMed (publishedVersion)
- Research at the University of Copenhagen (University of Copenhagen) (publishedVersion, other-oa)
- Scopus (Elsevier) (submittedVersion)
- Edinburgh Research Explorer (University of Edinburgh) (submittedVersion, cc-by-nd)
- St Andrews Research Repository (St Andrews Research Repository) (submittedVersion)
- Funders
- Ferring Pharmaceuticals
- Merck
- Novo Nordisk Fonden
- ReproUnion
- Fundacja na rzecz Nauki Polskiej
- Topics
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Reproductive Biology and Fertility
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Clinical Aspects of Sex Determination and Chromosomal Abnormalities
- Health Sciences › Medicine › Reproductive Medicine › Ovarian function and disorders
- Keywords
- Turner syndrome, ovarian tissue cryopreservation, ovarian follicles, follicle morphology, apoptosis, zona pellucida proteins, anti-Müllerian hormone, ovarian reserve, mosaic Turner syndrome
- MeSH terms
- Clinical Decision-Making, Adolescent, Adult, Biopsy, Child, Child, Preschool, Female, Fertility, Ovarian Follicle, Humans, Infant, Infertility, Female, Ovary, Phenotype, Predictive Value of Tests, Retrospective Studies, Turner Syndrome, Follicular Fluid, Cryopreservation, Patient Selection, Genetic Predisposition to Disease, Reproductive Techniques, Assisted, Young Adult, Fertility Preservation, In Vitro Oocyte Maturation Techniques
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