Hypothalamic-pituitary-gonadal function, pubertal development, and fertility outcomes in male and female medulloblastoma survivors: a single-center experience.
childhood cancer survivors
cyclophosphamide-equivalent dose
fertility
medulloblastoma
puberty
Journal
Neuro-oncology
ISSN: 1523-5866
Titre abrégé: Neuro Oncol
Pays: England
ID NLM: 100887420
Informations de publication
Date de publication:
06 Jul 2023
06 Jul 2023
Historique:
pmc-release:
12
01
2024
medline:
10
7
2023
pubmed:
13
1
2023
entrez:
12
1
2023
Statut:
ppublish
Résumé
Endocrine deficiencies, including hypothalamic-pituitary-gonadal axis (HPGA) impairment, are common in survivors of childhood and adolescent medulloblastoma. Still, data regarding pubertal development and fecundity are limited, and few studies assessed HPGA function in males. We aimed to describe HPGA function in a large cohort of patients with medulloblastoma. A retrospective study comprising all 62 medulloblastoma patients treated in our center between 1987 and 2021, who were at least 2 years from completion of therapy. HPGA function was assessed based on clinical data, biochemical markers, and questionnaires. Overall, 76% of female patients had clinical or biochemical evidence of HPGA dysfunction. Biochemical evidence of diminished ovarian reserve was seen in all prepubertal girls (n = 4). Among the males, 34% had clinical or biochemical evidence of gonadal dysfunction, 34% had normal function, and 29% were age-appropriately clinically and biochemically prepubertal. The difference between males and females was significant (P = .003). Cyclophosphamide-equivalent dose was significantly associated with HPGA function in females, but not in males. There was no association between HPGA dysfunction and other endocrine deficiencies, length of follow-up, weight status, and radiation treatment protocol. Two female and 2 male patients achieved successful pregnancies, resulting in 6 live births. HPGA dysfunction is common after treatment for childhood medulloblastoma. This is seen more in females, likely due to damage to the ovaries from spinal radiotherapy. Our findings may assist in counseling patients and their families regarding risk to future fertility and need for fertility preservation.
Sections du résumé
BACKGROUND
BACKGROUND
Endocrine deficiencies, including hypothalamic-pituitary-gonadal axis (HPGA) impairment, are common in survivors of childhood and adolescent medulloblastoma. Still, data regarding pubertal development and fecundity are limited, and few studies assessed HPGA function in males. We aimed to describe HPGA function in a large cohort of patients with medulloblastoma.
METHODS
METHODS
A retrospective study comprising all 62 medulloblastoma patients treated in our center between 1987 and 2021, who were at least 2 years from completion of therapy. HPGA function was assessed based on clinical data, biochemical markers, and questionnaires.
RESULTS
RESULTS
Overall, 76% of female patients had clinical or biochemical evidence of HPGA dysfunction. Biochemical evidence of diminished ovarian reserve was seen in all prepubertal girls (n = 4). Among the males, 34% had clinical or biochemical evidence of gonadal dysfunction, 34% had normal function, and 29% were age-appropriately clinically and biochemically prepubertal. The difference between males and females was significant (P = .003). Cyclophosphamide-equivalent dose was significantly associated with HPGA function in females, but not in males. There was no association between HPGA dysfunction and other endocrine deficiencies, length of follow-up, weight status, and radiation treatment protocol. Two female and 2 male patients achieved successful pregnancies, resulting in 6 live births.
CONCLUSIONS
CONCLUSIONS
HPGA dysfunction is common after treatment for childhood medulloblastoma. This is seen more in females, likely due to damage to the ovaries from spinal radiotherapy. Our findings may assist in counseling patients and their families regarding risk to future fertility and need for fertility preservation.
Identifiants
pubmed: 36633935
pii: 6986367
doi: 10.1093/neuonc/noad009
pmc: PMC10326472
doi:
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
1345-1354Informations de copyright
© The Author(s) 2023. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.
Références
Cancer Med. 2020 Aug;9(16):5807-5818
pubmed: 32608158
J Clin Endocrinol Metab. 2022 Apr 19;107(5):e2156-e2166
pubmed: 34918112
J Neurosurg. 1979 Jun;50(6):721-4
pubmed: 438873
Hum Reprod Update. 2022 May 2;28(3):417-434
pubmed: 35199161
J Pediatr. 2011 Jun;158(6):1016-1022.e1
pubmed: 21168856
J Pediatr. 1983 Oct;103(4):562-5
pubmed: 6620016
Med Pediatr Oncol. 2003 Apr;40(4):224-9
pubmed: 12555249
J Neurooncol. 2009 Nov;95(2):271-279
pubmed: 19521664
Hum Reprod Update. 2020 Nov 1;26(6):874-885
pubmed: 32935838
JMIR Res Protoc. 2018 Sep 14;7(9):e10824
pubmed: 30215599
Childs Nerv Syst. 2019 Dec;35(12):2327-2338
pubmed: 31686139
Neuro Oncol. 2011 Jun;13(6):669-79
pubmed: 21636711
Lancet Oncol. 2016 May;17(5):567-76
pubmed: 27020005
Rev Chil Pediatr. 2019 Dec;90(6):598-605
pubmed: 32186582
Pediatr Blood Cancer. 2015 Feb;62(2):329-334
pubmed: 25327609
Pediatr Blood Cancer. 2015 Feb;62(2):317-321
pubmed: 25346052
Obstet Gynecol Int. 2012;2012:564794
pubmed: 22619680
Pediatr Blood Cancer. 2014 Jan;61(1):53-67
pubmed: 23940101
J Clin Oncol. 2009 May 10;27(14):2374-81
pubmed: 19364956
Int J Radiat Oncol Biol Phys. 1986 Oct;12(10):1771-7
pubmed: 3759529
J Clin Endocrinol Metab. 2017 Mar 01;102(3):709-757
pubmed: 28359099
Childs Brain. 1981;8(2):107-18
pubmed: 7018855
Neuro Oncol. 2004 Apr;6(2):113-8
pubmed: 15134625
Arch Dis Child. 1988 May;63(5):495-500
pubmed: 3389863
J Clin Oncol. 2019 Mar 20;37(9):731-740
pubmed: 30730781
Am J Manag Care. 2016 Jun;22(7 Suppl):s176-85
pubmed: 27356115
Arch Dis Child. 1983 Sep;58(9):722-7
pubmed: 6414386
Sci Rep. 2020 Jun 10;10(1):9371
pubmed: 32523021
Hum Reprod. 2021 May 17;36(6):1561-1573
pubmed: 33744927
J Clin Endocrinol Metab. 1982 Jun;54(6):1164-8
pubmed: 6804477
J Neurooncol. 2022 Sep;159(2):243-259
pubmed: 35864412
Neuro Oncol. 2016 Jun;18(6):881-7
pubmed: 26688075
J Clin Endocrinol Metab. 2017 Jul 1;102(7):2242-2250
pubmed: 28368472
Clin Endocrinol (Oxf). 2015 Nov;83(5):663-70
pubmed: 25952583