Testicular biopsy for fertility preservation in early-diagnosed Klinefelter patients: patient characteristics and long-term follow-up

Aude Braye; Susanne Bohler; Veerle Vloeberghs; Veerle Rita J De Boe; Jean De Schepper; Inge Gies; Ellen Goossens

doi:10.1016/j.rbmo.2022.01.014

Testicular biopsy for fertility preservation in early-diagnosed Klinefelter patients: patient characteristics and long-term follow-up

Aude Braye, Susanne Bohler, Veerle Vloeberghs, Veerle Rita J De Boe, Jean De Schepper, Inge Gies, Ellen Goossens

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Abstract

Research question: Which early-diagnosed Klinefelter syndrome patients have been offered cryopreservation of testicular tissue as part of fertility preservation before spermatogonial stem cell (SSC) loss? Do these Klinefelter syndrome patients present with behavioural, cognitive and/or psychological problems? Does a testicular biopsy procedure have long-term effects on the gonadal development of Klinefelter syndrome patients? Design: Early-diagnosed Klinefelter syndrome patients followed between 2009 and 2020 and offered testicular tissue banking in an experimental context at the Universitair Ziekenhuis Brussel were included. The prevalence of behavioural, cognitive and/or psychological problems was determined. Changes in testicular volume and in gonadal function (LH, FSH, testosterone and inhibin B [INHB]) were studied. Results: Of the 48 Klinefelter syndrome patients included, 22 had testicular tissue removed (biopsy group) and 26 had no surgical intervention (control group). The need for specialized education was significantly higher in prenatally (P = 0.0159) and prepubertally (P = 0.0002) diagnosed Klinefelter syndrome patients. Psychological problems were significantly more prevalent in Klinefelter syndrome patients who did not opt for fertility preservation (P = 0.0447). In the first 4.2 (1.9–9.1) years after testicular biopsy, no difference in testicular volume was observed between the biopsied and the contralateral non-biopsied testes (P > 0.9999). After pubertal onset, no differences in LH, FSH, testosterone and INHB were found between the biopsy and the control groups (P = 0.1324 for LH, P > 0.9999 for FSH, P = 0.5433 for testosterone, P > 0.9999 for INHB). Conclusion: Early-diagnosed Klinefelter syndrome patients presented with behavioural, cognitive and/or psychological problems. Only psychological problems seemed to influence the decision towards fertility preservation. Follow-up data confirm that harvesting testicular tissue does not have a long-term impact on the gonadal development of Klinefelter syndrome patients.

Original language	English
Pages (from-to)	889-895
Number of pages	7
Journal	Reproductive BioMedicine Online
Volume	44
Issue number	5
DOIs	https://doi.org/10.1016/j.rbmo.2022.01.014
Publication status	Published - May 2022

Bibliographical note

Funding Information:
The authors are thankful to the Klinefelter Clinic of the UZ Brussel, E Van Moer (study nurse at the UZ Brussel fertility clinic) and M De Henau (medicine student at the VUB) for their help with data collection. This retrospective cohort study was supported by the Vrije Universiteit Brussel and Wetenschappelijk Fonds Willy Gepts of the UZ Brussel. The funding organizations had no role in the study conception and design, data collection, statistical analysis and interpretation of the results, supervision of the research and validation of the results, preparation, review or approval of the manuscript or the decision to submit the manuscript for publication.

Funding Information:
This retrospective cohort study was supported by the Vrije Universiteit Brussel and Wetenschappelijk Fonds Willy Gepts of the UZ Brussel. The funding organizations had no role in the study conception and design, data collection, statistical analysis and interpretation of the results, supervision of the research and validation of the results, preparation, review or approval of the manuscript or the decision to submit the manuscript for publication.

Publisher Copyright:
© 2022 Reproductive Healthcare Ltd.

Keywords

Fertility preservation
Gonadal development
Klinefelter syndrome
Retrospective cohort study
Testicular biopsy

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10.1016/j.rbmo.2022.01.014

1-s2.0-S1472648322000748-mainAccepted author manuscript, 1.09 MBLicence: CC BY-NC-ND

Cite this

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title = "Testicular biopsy for fertility preservation in early-diagnosed Klinefelter patients: patient characteristics and long-term follow-up",

abstract = "Research question: Which early-diagnosed Klinefelter syndrome patients have been offered cryopreservation of testicular tissue as part of fertility preservation before spermatogonial stem cell (SSC) loss? Do these Klinefelter syndrome patients present with behavioural, cognitive and/or psychological problems? Does a testicular biopsy procedure have long-term effects on the gonadal development of Klinefelter syndrome patients? Design: Early-diagnosed Klinefelter syndrome patients followed between 2009 and 2020 and offered testicular tissue banking in an experimental context at the Universitair Ziekenhuis Brussel were included. The prevalence of behavioural, cognitive and/or psychological problems was determined. Changes in testicular volume and in gonadal function (LH, FSH, testosterone and inhibin B [INHB]) were studied. Results: Of the 48 Klinefelter syndrome patients included, 22 had testicular tissue removed (biopsy group) and 26 had no surgical intervention (control group). The need for specialized education was significantly higher in prenatally (P = 0.0159) and prepubertally (P = 0.0002) diagnosed Klinefelter syndrome patients. Psychological problems were significantly more prevalent in Klinefelter syndrome patients who did not opt for fertility preservation (P = 0.0447). In the first 4.2 (1.9–9.1) years after testicular biopsy, no difference in testicular volume was observed between the biopsied and the contralateral non-biopsied testes (P > 0.9999). After pubertal onset, no differences in LH, FSH, testosterone and INHB were found between the biopsy and the control groups (P = 0.1324 for LH, P > 0.9999 for FSH, P = 0.5433 for testosterone, P > 0.9999 for INHB). Conclusion: Early-diagnosed Klinefelter syndrome patients presented with behavioural, cognitive and/or psychological problems. Only psychological problems seemed to influence the decision towards fertility preservation. Follow-up data confirm that harvesting testicular tissue does not have a long-term impact on the gonadal development of Klinefelter syndrome patients.",

keywords = "Fertility preservation, Gonadal development, Klinefelter syndrome, Retrospective cohort study, Testicular biopsy",

author = "Aude Braye and Susanne Bohler and Veerle Vloeberghs and {De Boe}, {Veerle Rita J} and {De Schepper}, Jean and Inge Gies and Ellen Goossens",

note = "Funding Information: The authors are thankful to the Klinefelter Clinic of the UZ Brussel, E Van Moer (study nurse at the UZ Brussel fertility clinic) and M De Henau (medicine student at the VUB) for their help with data collection. This retrospective cohort study was supported by the Vrije Universiteit Brussel and Wetenschappelijk Fonds Willy Gepts of the UZ Brussel. The funding organizations had no role in the study conception and design, data collection, statistical analysis and interpretation of the results, supervision of the research and validation of the results, preparation, review or approval of the manuscript or the decision to submit the manuscript for publication. Funding Information: This retrospective cohort study was supported by the Vrije Universiteit Brussel and Wetenschappelijk Fonds Willy Gepts of the UZ Brussel. The funding organizations had no role in the study conception and design, data collection, statistical analysis and interpretation of the results, supervision of the research and validation of the results, preparation, review or approval of the manuscript or the decision to submit the manuscript for publication. Publisher Copyright: {\textcopyright} 2022 Reproductive Healthcare Ltd.",

year = "2022",

month = may,

doi = "10.1016/j.rbmo.2022.01.014",

language = "English",

volume = "44",

pages = "889--895",

journal = "Reproductive BioMedicine Online",

issn = "1472-6483",

publisher = "Elsevier",

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Testicular biopsy for fertility preservation in early-diagnosed Klinefelter patients: patient characteristics and long-term follow-up. / Braye, Aude; Bohler, Susanne; Vloeberghs, Veerle ; De Boe, Veerle Rita J ; De Schepper, Jean ; Gies, Inge ; Goossens, Ellen.

In: Reproductive BioMedicine Online, Vol. 44, No. 5, 05.2022, p. 889-895.

Research output: Contribution to journal › Article › peer-review

TY - JOUR

T1 - Testicular biopsy for fertility preservation in early-diagnosed Klinefelter patients: patient characteristics and long-term follow-up

AU - Braye, Aude

AU - Bohler, Susanne

AU - Vloeberghs, Veerle

AU - De Boe, Veerle Rita J

AU - De Schepper, Jean

AU - Gies, Inge

AU - Goossens, Ellen

N1 - Funding Information: The authors are thankful to the Klinefelter Clinic of the UZ Brussel, E Van Moer (study nurse at the UZ Brussel fertility clinic) and M De Henau (medicine student at the VUB) for their help with data collection. This retrospective cohort study was supported by the Vrije Universiteit Brussel and Wetenschappelijk Fonds Willy Gepts of the UZ Brussel. The funding organizations had no role in the study conception and design, data collection, statistical analysis and interpretation of the results, supervision of the research and validation of the results, preparation, review or approval of the manuscript or the decision to submit the manuscript for publication. Funding Information: This retrospective cohort study was supported by the Vrije Universiteit Brussel and Wetenschappelijk Fonds Willy Gepts of the UZ Brussel. The funding organizations had no role in the study conception and design, data collection, statistical analysis and interpretation of the results, supervision of the research and validation of the results, preparation, review or approval of the manuscript or the decision to submit the manuscript for publication. Publisher Copyright: © 2022 Reproductive Healthcare Ltd.

PY - 2022/5

Y1 - 2022/5

N2 - Research question: Which early-diagnosed Klinefelter syndrome patients have been offered cryopreservation of testicular tissue as part of fertility preservation before spermatogonial stem cell (SSC) loss? Do these Klinefelter syndrome patients present with behavioural, cognitive and/or psychological problems? Does a testicular biopsy procedure have long-term effects on the gonadal development of Klinefelter syndrome patients? Design: Early-diagnosed Klinefelter syndrome patients followed between 2009 and 2020 and offered testicular tissue banking in an experimental context at the Universitair Ziekenhuis Brussel were included. The prevalence of behavioural, cognitive and/or psychological problems was determined. Changes in testicular volume and in gonadal function (LH, FSH, testosterone and inhibin B [INHB]) were studied. Results: Of the 48 Klinefelter syndrome patients included, 22 had testicular tissue removed (biopsy group) and 26 had no surgical intervention (control group). The need for specialized education was significantly higher in prenatally (P = 0.0159) and prepubertally (P = 0.0002) diagnosed Klinefelter syndrome patients. Psychological problems were significantly more prevalent in Klinefelter syndrome patients who did not opt for fertility preservation (P = 0.0447). In the first 4.2 (1.9–9.1) years after testicular biopsy, no difference in testicular volume was observed between the biopsied and the contralateral non-biopsied testes (P > 0.9999). After pubertal onset, no differences in LH, FSH, testosterone and INHB were found between the biopsy and the control groups (P = 0.1324 for LH, P > 0.9999 for FSH, P = 0.5433 for testosterone, P > 0.9999 for INHB). Conclusion: Early-diagnosed Klinefelter syndrome patients presented with behavioural, cognitive and/or psychological problems. Only psychological problems seemed to influence the decision towards fertility preservation. Follow-up data confirm that harvesting testicular tissue does not have a long-term impact on the gonadal development of Klinefelter syndrome patients.

AB - Research question: Which early-diagnosed Klinefelter syndrome patients have been offered cryopreservation of testicular tissue as part of fertility preservation before spermatogonial stem cell (SSC) loss? Do these Klinefelter syndrome patients present with behavioural, cognitive and/or psychological problems? Does a testicular biopsy procedure have long-term effects on the gonadal development of Klinefelter syndrome patients? Design: Early-diagnosed Klinefelter syndrome patients followed between 2009 and 2020 and offered testicular tissue banking in an experimental context at the Universitair Ziekenhuis Brussel were included. The prevalence of behavioural, cognitive and/or psychological problems was determined. Changes in testicular volume and in gonadal function (LH, FSH, testosterone and inhibin B [INHB]) were studied. Results: Of the 48 Klinefelter syndrome patients included, 22 had testicular tissue removed (biopsy group) and 26 had no surgical intervention (control group). The need for specialized education was significantly higher in prenatally (P = 0.0159) and prepubertally (P = 0.0002) diagnosed Klinefelter syndrome patients. Psychological problems were significantly more prevalent in Klinefelter syndrome patients who did not opt for fertility preservation (P = 0.0447). In the first 4.2 (1.9–9.1) years after testicular biopsy, no difference in testicular volume was observed between the biopsied and the contralateral non-biopsied testes (P > 0.9999). After pubertal onset, no differences in LH, FSH, testosterone and INHB were found between the biopsy and the control groups (P = 0.1324 for LH, P > 0.9999 for FSH, P = 0.5433 for testosterone, P > 0.9999 for INHB). Conclusion: Early-diagnosed Klinefelter syndrome patients presented with behavioural, cognitive and/or psychological problems. Only psychological problems seemed to influence the decision towards fertility preservation. Follow-up data confirm that harvesting testicular tissue does not have a long-term impact on the gonadal development of Klinefelter syndrome patients.

KW - Fertility preservation

KW - Gonadal development

KW - Klinefelter syndrome

KW - Retrospective cohort study

KW - Testicular biopsy

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DO - 10.1016/j.rbmo.2022.01.014

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JO - Reproductive BioMedicine Online

JF - Reproductive BioMedicine Online

SN - 1472-6483

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ER -

Testicular biopsy for fertility preservation in early-diagnosed Klinefelter patients: patient characteristics and long-term follow-up

Abstract

Bibliographical note

Keywords

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FWOAL1023: Fertility preservation in boys treated for cancer: evaluating the impact of harvesting testicular tissue at young age on the later pubertal development and fertility (prospective study)

FWOAL891: Fertility preservation in boys treated for cancer: what is the effect of harvesting testicular tissue in childhood on their later pubertal development and fertility?

Cite this

Testicular biopsy for fertility preservation in early-diagnosed Klinefelter patients: patient characteristics and long-term follow-up

Abstract

Bibliographical note

Keywords

Access to Document

Other files and links

Fingerprint

Projects

FWOAL1023: Fertility preservation in boys treated for cancer: evaluating the impact of harvesting testicular tissue at young age on the later pubertal development and fertility (prospective study)

FWOAL891: Fertility preservation in boys treated for cancer: what is the effect of harvesting testicular tissue in childhood on their later pubertal development and fertility?

Cite this