Antisperm antibodies in the polyglandular autoimmune (PGA) syndrome type I: response to cyclical steroid therapy.

Abstract

OBJECTIVE: To determine if cyclical intermediate dose steroid therapy could improve semen parameters in an infertile man with sperm autoimmunity associated with the polyglandular autoimmune (PGA) syndrome. DESIGN: Sperm agglutination studies performed before, during and after three courses of cyclical intermediate dose prednisolone therapy. PATIENT: A twenty-six-year old man with polyglandular autoimmune syndrome, consisting of Addison's disease, hypoparathyroidism, chronic mucocutaneous candidiasis and alopecia totalis, presented with infertility. He had normal endocrine testicular function but severe exocrine failure evidenced by a low sperm count (4.5 x 10(6)/ml), zero motility and universal sperm agglutination. MEASUREMENTS: Sperm agglutination tests. RESULTS: At presentation the gelatin agglutination test (GAT) was strongly positive in serum (1/1204) and seminal plasma (1/64) as was the tray agglutination test (TAT) (1/32). The patient's wife had a regular menstrual cycle with normal luteal phase progesterone levels. Following three courses of cyclical prednisolone (20 mg twice daily on days 1-10 of wife's cycle, and 5 mg on days 11 and 12), sperm quantity and motility improved considerably (12 x 10(6)/ml, 40% respectively) and sperm agglutination tests became negative. After a fourth course of therapy the patient's wife became pregnant. Three months post-treatment sperm motility was very low again and agglutinating activity in serum and seminal plasma increased. CONCLUSIONS: This is the first case of male infertility due to sperm autoimmunity in association with the PGA syndrome type 1. The immunosuppressive action of cyclical intermediate dose steroid therapy led to a significant quantitative and qualitative improvement in semen parameters.