Adrenogenital syndrome: unusual presentation in an adult

A case of bilateral testicular tumors thought secondary to hyperplastic adrenal rest tissue in an adult with the adrenogenital syndrome is presented. The appropriate endocrinologic diagnosis of 21-hydroxylase deficiency was made by extensive hormonal assay of serum and urine. Pathologic investigation suggested Leydig cell hyperplasia or tumor, but clinical and laboratory response to steroid therapy indicated elaboration of androgens from adrenal or adrenal rest sources. We believe initial medical therapy is justified in these particular cases.