ESPE2015 Poster Presentations Poster Category 1 DSD (11 abstracts)
Subcutaneous Continuous Administration of Recombinant Human Luteinizing and Follicle-Stimulating Hormones is an Effective Treatment for Micropenis During the Mini-Puberty
Athanasia Stoupa a , Dinane Samara-Boustani a, , Isabelle Flechtner a, , Graziella Pinto a, , Isabelle Jourdon a , Kathleen Laborde c , Didier Chevenne d , Anne-Elodie Millischer-Bellaiche e , Michel Polak a, & Jacques Beltrand a,
aEndocrinologie, Gynécologie et Diabétologie Pédiatriques, Hôpital Universitaire Necker Enfants Malades, Assistance Publique-Hôpitaux de Paris, Paris, France; bCentre des Maladies Endocriniennes Rares de la Croissance, Hôpital Universitaire Necker Enfants Malades, Assistance Publique-Hôpitaux de Paris, Paris, France; cExplorations Fonctionnelles, Hôpital Universitaire Necker Enfants Malades, Assistance Publique-Hôpitaux de Paris, Paris, France; dBiochimie et Hormonologie, Hôpital Universitaire Robert Debré, Assistance Publique-Hôpitaux de Paris, Paris, France; eRadiologie Pédiatrique, Hôpital Universitaire Necker Enfants Malades, Assistance Publique-Hôpitaux de Paris, Paris, France; fINSERM U1016, IMAGINE Institute, Université Paris Descartes, Sorbonne Paris Cité, Paris, France.
Background: Early postnatal administration of recombinant human gonadotropins can be an effective way to mimic mini-puberty, and thus increase penile growth in infants with congenital hypogonadotropic hypogonadism (CHH). We report for the first time its efficacy on an infant with partial androgen insensitivity syndrome (PAIS).
Objective and hypotheses: To evaluate the benefits of a continuous subcutaneous infusion of recombinant human gonadotropins (CSCI-HGon) on penile length and hormonal response, in a group of infants with micropenis and cryptorchidism.
Method: Six male patients (isolated CHH, n=4, panhypopituitarism, n=1, PAIS, n=1) were treated at a mean age of 3.8 months with continuous recombinant LH (Luveris, Merck Serono) and FSH (Gonal-F, Merck Serono) delivered via a pump. Clinical (stretched penile length (SPL), testicular position and volume), hormonal (testosterone, LH, FSH, anti-Mullerian hormone, AMH and inhibin B) and radiological (testicular position and size) parameters were evaluated at baseline, during and at the end of the treatment.
Results: CSCI-HGon was administered during a mean duration of 4.3 months. In CHH patients, CSCI-HGon increased serum testosterone concentration at 3.5±4.06 ng/dl, and thus, SPL (from 13.8±4.5 to 42.6±5 mm, P<0.0001). At the end of the treatment, micropenis was corrected in all patients, except one, who therefore received intramuscular testosterone. Inhibin B (from 94.8±74.9 pg/ml to 469.4±282.5 pg/l, P=0.04) and AMH (from 49.6±30.6 to 142±76.5 ng/ml, P=0.03) also increased, reflecting the efficacy of the treatment on Sertoli cells. For PAIS patient, final SPL significantly increased (from 13 to 38 mm) under higher doses of LH.
Conclusion: Early CSI-HGon is an effective and safe treatment, mimicking the physiological neonatal activation of gonadotropic axis restoring a normal penile length. Furthermore, we report for the first time its effectiveness at a patient with PAIS. Long-term follow-up is needed in order to evaluate the consequences in future fertility and reproductive function.
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