Endocrine Connections (Mar 2025)

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

  • Anne-Sophie Lambert,
  • Marie-Agathe Trouvin,
  • Khadidja Fouatih,
  • Adrien Lecoeuvre,
  • Cécile Thomas-Teinturier,
  • Anya Rothenbuhler,
  • Jérôme Bouligand,
  • Severine Trabado,
  • Agnès Linglart,
  • Claire Bouvattier

DOI
https://doi.org/10.1530/EC-24-0082
Journal volume & issue
Vol. 14, no. 4

Abstract

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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.

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