Successful treatment with ICSI of infertility caused by azoospermia associated with adrenal rests in the testes

Congenital adrenal hyperplasia (CAH) is a well-recognized, but uncommon, ca use of azoospermia and infertility in men. Commonly this is due to undertre atment of excessive adrenal androgen secretion which suppresses gonadotroph in stimulation of the testes. A less common complication of CAH is developm ent of adrenal tissue within the testes; this is important to recognize bec ause it may be confused with malignancy leading to unnecessary surgery. In this case report, a man is described with simple virilizing CAH due to 21-h ydroxylase deficiency who presented with azoospermia and was found to have adrenal rests. Investigations concluded that there was adequate adrenal sup pression with glucocorticoids and that azoospermia was due to obstruction b y adrenal rest tissue, strategically situated at the hilum of the testes. S permatozoa were able to be retrieved by testicular aspiration from the man and these were used to successfully establish a pregnancy using intracytopl asmic sperm injection of his wife's oocytes.