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Primary Gonadal Insufficiency in Male and Female Childhood Cancer Survivors in a Long-Term Follow-Up Clinic
Childrens Hospital Westmead, Australia; University of Sydney, Australia.
Linköping University, Faculty of Medicine and Health Sciences.
Linköping University, Faculty of Medicine and Health Sciences.
Childrens Hospital Westmead, Australia.
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2016 (English)In: Journal of Adolescent and Young Adult Oncology, ISSN 2156-5333, Vol. 5, no 4, 344-350 p.Article in journal (Refereed) Published
Abstract [en]

Purpose: Childhood cancer survivors (CCS) are at increased risk of primary gonadal insufficiency (PGI). This study evaluated the prevalence and clinical characteristics of PGI in CCS. Methods: In this single-center, retrospective, observational, longitudinal study, we characterized CCS with PGI attending the oncology Long-Term Follow-Up (LTFU) Clinic at an Australian university hospital (January 2012-August 2014). From a cohort of 276 CCS, 54 (32 males) met criteria for PGI: elevated gonadotropins plus low estradiol/amenorrhoea (females) or low testosterone/small testicles for age (males). Results: Median age at primary diagnosis was 4.8 years (inter-quartile range [IQR] 3.0-9.7 years) and at LTFU, it was 22.3 years (IQR 18.2-25.7 years). Fifty-three participants (98.1%) were treated with known highly gonadotoxic therapies: alkylating chemotherapy (96.3%), radiotherapy (70.3%), total body irradiation (29.6%), bone marrow transplantation (51.9%), or multimodal protocols (68.5%). At primary diagnosis, 86.7% participants were Tanner stage I and at LTFU, 89.1% participants were Tanner stage V. More females (95.5%; n=21) than males (40.6%; n=13) were treated with hormone development therapy (HDT) (pamp;lt;0.01). Of these, more than half (n=18; 7 males) required pubertal induction. There was no significant difference in serum luteinizing hormone/follicle stimulating hormone (LH/FSH), testosterone/estradiol between those untreated and those treated with HDT. Among those on HDT, 60.7% had persistently elevated FSHLH and 33.3% had low testosterone or estradiol. Six males had semen analysis (five azoospermic, one oligospermic). Psychological assessment was documented in 61.1% of participants, and two-thirds reported fertility concerns. Conclusion: PGI is an evolving phenotype that is common in CCS. Suboptimal treatment and non-adherence occur frequently. Ongoing assessment is essential to ensure prompt diagnosis, adequate intervention and to promote HDT adherence.

Place, publisher, year, edition, pages
MARY ANN LIEBERT, INC , 2016. Vol. 5, no 4, 344-350 p.
Keyword [en]
primary gonadal insufficiency; gonadal failure; fertility; hormone therapy; late effects
National Category
Pediatrics
Identifiers
URN: urn:nbn:se:liu:diva-133889DOI: 10.1089/jayao.2016.0007ISI: 000390242200006PubMedID: 27195593OAI: oai:DiVA.org:liu-133889DiVA: diva2:1065085
Available from: 2017-01-13 Created: 2017-01-13 Last updated: 2017-01-13

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