Background: Childhood onset (CO) hypogonadotrophic hypogonadism (HH), congenital or acquired after midline tumours and their treatment, is reported to have significantly poorer outcomes, in terms of adult spermatogenesis induction and duration of treatment to conception, compared with HH of post pubertal onset. A mixed cohort (youngest aged 26 years) reported median time to conception of 28 months.1 Use of hCG and FSH in adolescence is reported to result in spermatogenesis after 69 months of combined treatment.2 Presence of other pituitary hormone deficiencies did not alter outcomes. Little data exist regarding fertility outcomes for this group. A recent report confirmed a poorer fertility outcome in men with CO HH, particularly Kallman syndrome, with absent spontaneous puberty, testicular size before treatment correlating with time to fertility, with 8 conceptions, 3/8 requiring ICSI. Birth rate was not reported.3
Objective and hypotheses: To report adult outcomes for a cohort of 14 men with congenital HH.
Method: Three had past pubertal induction with hCG/FSH, 11/14 used testosterone alone. Age at first adult use of hCG and FSH for fertility induction ranged from 2632 years. All achieved adult range testosterone levels within 34 months. Eight have sought fertility using FSH to date.
Results: Time to spermatogenesis for first induction: median 9 months (936 months), mean sperm numbers: 1.78×106 (1000-4×106). Time to first fertility: mean 28 months (951 months, IVF/ICSI needed in one), with live births in all. Second round fertility induction was undertaken by 3, mean time to sperm 3.5 months, time to fertility 8 months, all with live births. A total of 9 live normal births is reported.
Conclusion: These men with CO HH demonstrate similar characteristics to earlier reports, with more rapid onset of spermatogenesis in second round induction. Nine normal live births, for men with CO HH,only one needing ICSI is the largest cohort so far identified.
References: 1. Liu PY, Baker HW, Jayadev V, Zacharin M, Conway AJ, Handelsman DJ. J CEM 2009;94(3):8018. 2. Zacharin M1, Sabin MA, Nair VV, Dabadghao PAndrology. 2016;4(1):8794. 3. Rohayem J, Sinthofen N, Nieschlag E, Kliesch S, Zitzmann M. Fertil Steril. 2012;98(4):83642.
10 - 12 Sep 2016
European Society for Paediatric Endocrinology