Insulin-induced ovarian hyperstimulation and hyperandrogenization can cause virilization in children and adults. It rarely occurs in children and adolescents but when it does, it is usually caused by congenital adrenal hyperplasia or virilizing tumors. In very rare cases severe hyperandrogenism is caused by severe insulin resistance, either due to germline mutations in genes encoding the insulin receptor or proteins in downstream insulin signaling or due to insulin receptor antibodies
Case description: A 13-year-old adolescent girl with monogenic severe insulin resistance due to an INSR mutation encoding a substitution in the tyrosine kinase-encoding domain (P. Leu1150Arg) developed severe hyperandrogenism with virilization. Testosterone levels were elevated into the adult male range. Treatment with a gonadotropin-releasing hormone (GnRH) analog led to normalization of testosterone (and insulin C-peptide) and remission of the clinical findings, which persisted with the subsequent switch of treatment with an anti-androgenic contraceptive.
Conclusion: Suppression of gonadotropins with anti-androgenic contraceptives may be a feasible therapeutic modality in insulin-induced severe ovarian hyperstimulation and hyperandrogenism.
15 Sep 2022 - 17 Sep 2022