Abstract
We describe the case of a baby born to a mother with Addison’s disease in the context of Autoimmune Polyendocrine Syndrome Type 2._Adrenal cortex autoantibodies and steroid 21-hydroxylase autoantibodies were detectable in the sera of both mother and baby, suggesting the transplacental passage of these autoantibodies. Adrenal autoantibodies were present in the baby’s serum at delivery, at 3, 6 and till 34 months of age but no signs of clinical or subclinical adrenal insufficiency were found in the baby during the observation period. These data suggest that the presence of adrenal autoantibodies in serum alone is not a sufficient cause for the development of autoimmune adrenalitis.
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Betterle C, Dal Pra C, Mantero F, Zanchetta R. Autoimmune adrenal insufficiency and autoimmnune polyendocrine syndromes: autoantibodies, autoantigens and their applicability in diagnosis and disease prediction. Endocr Rev 2002, 23: 327–64.
Van der Spuy ZM, Jacobs HS. Management of endocrine disorders in pregnancy. Part II-pituitary, ovarian and adrenal diseases. Postgrad Med J 1984, 60: 312–20.
Cowchock FS, McCabe JL, Montgomery BB. Pregnancy after corticosteroid administration in premature ovarian failure (polyglandular endocrinopathy syndrome). Am J Obstet Gynecol 1988, 158: 118–19.
Seaward PGR, Guidozzi F, Sonnendecker EWW. Addisonian crisis in pregnancy. Case report. Brit J Obstet Gynaecol 1989, 96: 1348–50.
Albert E, Dalaker K, Jorde R, Berge LN. Addison’s disease and pregnancy. Acta Obstect Gyn Scan 1989, 68: 185–7.
George LD, Selvaraju R, Reddy K, Stout TV, Premawardhana LDKE. Vomiting and hyponatriemia in pregancy. Brit J Obstet Gynaecol 2000, 107: 808–9.
Anderson JR, Goudie RB, Gray KG, Timbury GC. Autoantibodies in Addison’s disease. Lancet 1957, 1: 1123–24.
Baumann-Antczak A, Wedlock N, Bednarek J, et al. Autoimmune Addison’s disease and 21-hydroxylase. Lancet 1992, 340: 429–30.
Winqvist O, Karlsson FA, Kämpe O. 21-hydroxylase, a major autoantigen in idiopathic Addison’s disease. Lancet 1992, 339: 1559–62.
Furmaniak J, Kominami S, Asawa T, Wedlock N, Colls J, Rees Smith B. Autoimmune Addison’s disease. Evidence for a role of steroid 21-hydroxylase autoantibodies in adrenal insufficiency. J Clin Endocrinol Metab 1994, 79: 1517–21.
Boscaro M, Betterle C, Volpato M, et al. Hormonal responses during various phases of autoimmune adrenal failure: no evidence for 21-hydroxylase enzyme activity inhibition in vivo. J Clin Endocrinol Metab 1996, 81: 2801–4.
Irvine WJ, Stewart AG, Scarth L. A clinical and immunological study of adrenocortical insufficiency (Addison’s disease). Clin Exp Immunol 1967, 2: 31–70.
Baum JD, Chantler C. Hyperinsulinemic child of mother with Addison’s disease. Proc Roy Soc Med 1968, 61: 1261–2.
Nerup J. Addison’s disease-serological studies. Acta Endocrinol (Copenh) 1974, 76: 142–58.
Poonai A, Jelercic F, Pop-Lazic B. Pregancy with diabetes mellitus, Addison’s disease and hypothyroidism. Obstet Gynecol 1977, 49: 86s–88s.
Gamlen TR, Aynsley-Green A, Irvine WJ, McCallum CJ. Immunological studies in the neonate of a mother with Addison’s disease and diabetes mellitus. Clin Exp Immunol 1977, 28: 192–5.
Tanaka H, Perez MS, Pawell M, et al. Steroid 21-hydroxylase autoantibodies measurements with a new immunoprecipitation assay. J Clin Endocrinol Metab 1997, 82: 1440–6.
Betterle C, Volpato M, Pedini B, Chen S, Smith BR, Furmaniak J. Adrenal-cortex autoantibodies (ACA) and steroidproducing cells autoantibodies (StCA in patients with Addison’s disease: comparison between immunofluorescence and immunoprecipitation assays. J Clin Endocrinol Metab 1999, 84: 618–22.
Chen S, Sawicka S, Betterle C, et al. Autoantibodies to steroidogenic enzymes in Autoimmune Polyglandular Syndrome, Addison’s disease and Premature Ovarian Failure. J Clin Endocrinol Metab 1996, 81: 871–6.
Lahansky G, Saenger P, Fishman K, et al.: Normative data for adrenal steroidogenesis in a healthy pediatric population: age- and sex-related changes after adrenocorticotropin stimulation. J Clin Endcorinol Metab 73: 674–86, 1991.
Billington WD. The normal fetomaternal immune relationship. Baillieres Clin Obstet Gynecol 1992, 6: 417–38.
Zinkernagel RM. Maternal antibodies, childhood infections, and autoimmune diseases. N Engl J Med 2001, 345: 1331–5.
Matsuura N, Yamada Y, Nohara Y, et al. Familial neonatal transient hypothyroidism due to maternal TSH-binding inhibitor immunoglobulins. N Engl J Med 1980, 303: 738–41.
Rees Smith B, McLachlan SM, Furmaniak J. Autoantibodies to the thyrotropin receptor. Endocr Rev 1988, 9: 106–21.
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Betterle, C., Dal Pra, C., Pedini, B. et al. Assessment of adrenocortical function and autoantibodies in a baby born to a mother with autoimmune polyglandular syndrome Type 2. J Endocrinol Invest 27, 618–621 (2004). https://doi.org/10.1007/BF03347492
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DOI: https://doi.org/10.1007/BF03347492