Early adolescence assessment of eleven boys treated with continuous subcutaneous infusion of recombinant LH and FSH during infancy

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Abstract We previously observed that continuous subcutaneous gonadotropin infusion (CSGI) in infants with congenital hypogonadotropic hypogonadism (CHH) can mimic minipuberty. Objective To describe the early adolescence outcome of boys treated during the first year of life. Methods In this retrospective cohort study, we describe eleven boys with CHH aged 12 years (11.5–14.6) treated at the age of 4.5 months (2.0–11) with CSGI. As a retrospective control cohort, we report testicular function of 12 untreated boys with CHH aged 12 years (12–15.9). Results In response to CSGI, serum testosterone and inhibin B levels increased from 0.03 ng/mL (0–0.07) to 2.25 ng/mL (1.12–3.86) and from 73 (11–173) to 401 (185–727) pg/mL, respectively. The testicular volume increased from 0.50 mL (0.5–1) to 1.50 mL (0.7–3). Orchidopexy was performed in 4/11 patients. Between end of CSGI and early adolescence period, testicular volume in the treated group decreased from 1.5 mL (0.7–3) to 1.05 mL (0.7–2.36) (P = 0.024) and differed from that in untreated boys (0.3 mL (0.13–1.3); reference range (mL) for the Tanner stage (0.5–4.7)). Concerning hormonal status, hormone levels were higher in the treated group than in untreated group: serum AMH and inhibin B levels in treated patients decreased from 1028 pmol/L (550–1750) and 356 (185–727) pg/mL at neonatal period to 331 pmol/L (85–479) and 68 pg/mL (19–239), respectively, at early adolescence and differed from those in untreated patients (57.5 (30–169) and 8 pg/mL (<5–37), respectively (P < 0.001)). Conclusion We report the first long-term follow-up of boys with CHH treated with CSGI in infancy. Our results have shown that the CSGI treatment resulted in higher inhibin B, AMH levels and testicular volume, which had some persisting effects lasting until early adolescence age. Follow-up should be continued until the end of puberty to assess spermatogenesis. Significance statement Minipuberty is known to be of some importance for future fertility but is usually absent in infants with hypogonadotropic hypogonadism. CSGI treatment in minipuberty period can mimic this hormonal activation. We describe here the natural course at early adolescence of boys treated with CSGI during their first year of life. The effects shown in the study were higher inhibin B levels and larger testicular volume in patients treated by CSGI treatment, which had some persisting effects lasting until early adolescence period. Follow-up should be continued until the end of puberty to assess spermatogenesis in these patients.

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