Abstract
Congenital adrenal hyperplasia (CAH) causes hypogonadotropic hypogonadism due to the excessive production of adrenal androgens, which results in hypospermatogenesis in some male patients. We herein present a CAH case with hypogonadotropic hypogonadism and male infertility. A 26-year-old male receiving steroid therapy for 21 hydroxylase deficiency was diagnosed with low gonadotropin levels, an elevated ACTH level, and severe oligozoospermia. The switching from hydrocortisone to dexamethasone resulted in the normalization of gonadotropin levels and semen findings. The couple underwent ICSI-ET, resulting in a live birth. In cases of CAH with hypospermatogenesis, the continuous suppression of ACTH by dexamethasone may restore spermatogenesis.