Time course of 21-hydroxylase antibodies and long-term remission of subclinical autoimmune adrenalitis after corticosteroid therapy: case report.
Subclinical Addison's disease is characterized by the presence of adrenal autoantibodies (ACA) and steroid 21-hydroxylase autoantibodies (21OHAb) with or without adrenal function failure. In our previous longitudinal study some patients with high titers of ACA and at stage 2 of subclinical adrenocortical failure showed disappearance of ACA with recovery of normal adrenocortical function after corticosteroid treatment for Graves' ophthalmopathy. To investigate whether corticosteroid-induced modification of the adrenal autoimmune markers can also involve 21OHAb and to evaluate whether the remission of subclinical adrenocortical failure can persist over a long period of time, we followed-up for 100 months the levels of 21OHAb and ACA as well as the metabolic markers of adrenal function in one patient with Graves' ophthalmopathy and at stage 2 of subclinical adrenocortical failure before and after corticosteroid therapy. A 34-yr-old woman with Graves' disease and active ophthalmopathy who was found to be positive for ACA and to have high PRA, low aldosterone levels, and normal basal ACTH and cortisol levels, but impaired cortisol response to ACTH was studied. The patient was treated with oral corticosteroid therapy for 6 months. After corticosteroid therapy, 21OHAb, initially positive, became negative in concomitance with the disappearance of ACA and the restoration of normal adrenal function. The disappearance of both 21OHAb and ACA and their prolonged absence during the follow-up suggest that corticosteroid treatment can induce long-term remission of subclinical adrenal insufficiency and prevent the onset of the clinical phase of the disease. Our pilot study may pave the way to future trials aimed at preventing the onset of the clinical signs of Addison's disease in ACA/21OHAb-positive patients.
De Bellis A
A
A
Institute of Endocrinology and Department of Clinical and Experimental Medicine F. Magrassi, Second University of Naples, 80131 Naples, Italy. etxode@tin.it
Falorni
A
A
Laureti
S
S
Perrino
S
S
Coronella
C
C
Forini
F
F
Bizzarro
E
E
Bizzarro
A
A
Abbate
G
G
Bellastella
A
A
D000303:Adrenal Cortex Diseases / Q000097:blood / Q000139:chemically induced / Q000276:immunology*; D000305:Adrenal Cortex Hormones / Q000009:adverse effects*; D000324:Adrenocorticotropic Hormone / Q000097:blood; D000328:Adult; D000450:Aldosterone / Q000097:blood; D000906:Antibodies / Q000097:blood; D001327:Autoimmune Diseases / Q000097:blood / Q000139:chemically induced / Q000276:immunology*; D015415:Biomarkers / Q000097:blood; D005260:Female; D005500:Follow-Up Studies; D006111:Graves Disease / Q000188:drug therapy*; D006801:Humans; D006854:Hydrocortisone / Q000097:blood; D011241:Prednisone / Q000009:adverse effects*; D013255:Steroid 21-Hydroxylase / Q000276:immunology*; D013997:Time Factors
D000305:Adrenal Cortex Hormones; D000906:Antibodies; D015415:Biomarkers; D000450:Aldosterone; D000324:Adrenocorticotropic Hormone; D013255:Steroid 21-Hydroxylase; D011241:Prednisone; D006854:Hydrocortisone