PMID- 27195593
OWN - NLM
STAT- MEDLINE
DCOM- 20180130
LR  - 20180824
IS  - 2156-535X (Electronic)
IS  - 2156-5333 (Linking)
VI  - 5
IP  - 4
DP  - 2016 Dec
TI  - Primary Gonadal Insufficiency in Male and Female Childhood Cancer Survivors in a 
      Long-Term Follow-Up Clinic.
PG  - 344-350
AB  - 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) (p < 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 FSH+/-LH 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.
FAU - Gunn, Harriet M
AU  - Gunn HM
AD  - 1 Academic Department of Adolescent Medicine, The Children's Hospital at Westmead 
      , Sydney, New South Wales, Australia .
AD  - 2 Discipline of Child and Adolescent Health, The University of Sydney , Sydney, 
      New South Wales, Australia .
FAU - Rinne, Ida
AU  - Rinne I
AD  - 3 The Faculty of Medicine and Health Sciences, Linkoping University , Linkoping, 
      Sweden .
FAU - Emilsson, Hanna
AU  - Emilsson H
AD  - 3 The Faculty of Medicine and Health Sciences, Linkoping University , Linkoping, 
      Sweden .
FAU - Gabriel, Melissa
AU  - Gabriel M
AD  - 4 Long Term Follow Up Clinic, Department of Oncology, The Children's Hospital at 
      Westmead , Sydney, New South Wales, Australia .
FAU - Maguire, Ann M
AU  - Maguire AM
AD  - 2 Discipline of Child and Adolescent Health, The University of Sydney , Sydney, 
      New South Wales, Australia .
AD  - 5 Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead , 
      Sydney, New South Wales, Australia .
FAU - Steinbeck, Katharine S
AU  - Steinbeck KS
AD  - 1 Academic Department of Adolescent Medicine, The Children's Hospital at Westmead 
      , Sydney, New South Wales, Australia .
AD  - 2 Discipline of Child and Adolescent Health, The University of Sydney , Sydney, 
      New South Wales, Australia .
LA  - eng
PT  - Journal Article
DEP - 20160519
PL  - United States
TA  - J Adolesc Young Adult Oncol
JT  - Journal of adolescent and young adult oncology
JID - 101543508
SB  - IM
MH  - *Cancer Survivors
MH  - Child
MH  - Child, Preschool
MH  - Cohort Studies
MH  - Female
MH  - Follow-Up Studies
MH  - Humans
MH  - Hypogonadism/diagnosis/*etiology/physiopathology
MH  - Longitudinal Studies
MH  - Male
MH  - Prevalence
MH  - Retrospective Studies
OTO - NOTNLM
OT  - fertility
OT  - gonadal failure
OT  - hormone therapy
OT  - late effects
OT  - primary gonadal insufficiency
EDAT- 2016/05/20 06:00
MHDA- 2018/01/31 06:00
CRDT- 2016/05/20 06:00
PHST- 2016/05/20 06:00 [pubmed]
PHST- 2018/01/31 06:00 [medline]
PHST- 2016/05/20 06:00 [entrez]
AID - 10.1089/jayao.2016.0007 [doi]
PST - ppublish
SO  - J Adolesc Young Adult Oncol. 2016 Dec;5(4):344-350. doi: 10.1089/jayao.2016.0007. 
      Epub 2016 May 19.