Abstract A number of immunological abnormalities have been described in association with idiopathic hypoparathyroidism. These include the presence of circulating antibodies against various tissue antigens,1 abnormal invivo or in-vitro lymphocyte functions,2 and mucocutaneous fungal infections.3,4 In addition, a variety of endocrine and nonendocrine disorders occur in patients with idiopathic hypoparathyroidism,5 including hypoadrenalism,6,7 pernicious anemia,8,9 steatorrhea,10,11 and hypothyroidism.12 To these must be added yet another syndrome—primary amenorrhea or early ovarian failure. In this paper we describe the seventh such recorded case and briefly review the literature. Detailed immunological studies are presented and the association between cellular immune defects and endocrinological abnormalities is discussed. Patient Summary A girl was born on Oct 28, 1956, of unrelated parents after an uneventful pregnancy. Her father was 25 years of age and her mother 22 years at the time of her birth. The patient appeared normal in the References 1. Blizzard RM, Chee D, Davis W: The incidence of parathyroid and other antibodies in the sera of patients with idiopathic hypoparathyroidism. Clin Exp Immunol 1:119-128, 1966. 2. Hermans PE, Ulrich JA, Markowitz H: Chronic mucocutaneous candidiasis as a surface expression of deep-seated abnormalities. Am J Med 47:503-519, 1969.Crossref 3. Sutphin A, Albright F, McCune DJ: Five cases (three in siblings) of idiopathic hypoparathyroidism associated with moniliasis. J Clin Endocrinol 3:625-634, 1943.Crossref 4. Bronsky D, et al: Idiopathic hypoparathyroidism and pseudohypoparathyroidism: Case reports and review of the literature. Medicine 37:317-352, 1958.Crossref 5. Bronsky D: Hyperparathyroidism with Albright's osteodystrophy: Case report and a proposed new classification of parathyroid disease. J Clin Endocrinol Metab 31:271-276, 1970.Crossref 6. Talbot NB, Butler AM, McLachlan EA: The effect of testosterone and allied compounds on the mineral, nitrogen and carbohydrate metabolism of a girl with Addison's disease. J Clin Invest 22:583-593, 1943.Crossref 7. Craig JM, Schiff LH, Boone JF: Chronic moniliasis associated with Addison's disease. Am J Dis Child 89:669-684, 1955. 8. Hung W, Migeon CJ, Parrott RH: A possible autoimmune basis for Addison's disease in three siblings, one with idiopathic hypoparathyroidism, pernicious anemia and superficial moniliasis. N Engl J Med 269:658-663, 1963.Crossref 9. Kunin AS, et al: The syndrome of hypoparathyroidism and adrenocortical insufficiency, a possible sequel of hepatitis: Case report and review of the literature. Am J Med 34:856-866, 1963.Crossref 10. Whitaker JA, et al: The syndrome of familial juvenile hypoadrenocorticism, hypoparathyroidism and superficial moniliasis. J Clin Endocrinol Metab 16:1374-1387, 1956.Crossref 11. Morse WI, Cochrane WA, Landrigan PL: Familial hypoparathyroidism with pernicious anemia, steatorrhea and adrenocortical insufficiency. N Engl J Med 264:1021-1026, 1961.Crossref 12. Kenny FM, Holliday MA: Hypoparathyroidism, moniliasis, Addison's and Hashimoto's diseases: Hypercalcemia treated with intravenously administered sodium sulfate. N Engl J Med 271:708-713, 1964.Crossref 13. Ireland AW, et al: The calciferol requirements of patients with surgical hypoparathyroidism. Ann Intern Med 69:81-90, 1968.Crossref 14. Ireland AW, et al: The crystalline lens in chronic surgical hypoparathyroidism. Arch Intern Med 122:408-411, 1968.Crossref 15. Frensilli JA, Stoner RE, Hinrichs EH: Dental changes of idiopathic hypoparathyroidism: Report of three cases. J Oral Surg 29:727-731, 1971. 16. Greulich WW, Pyle SI: Radiographic Atlas of Skeletal Development of the Hand and Wrist , ed 2. Stanford, Calif, Stanford University Press, 1959. 17. Lehrer RI, Cline MJ: Interaction of Candida albicans with human leukocytes and serum. J Bacteriol 98:996-1004, 1969. 18. Fitzgerald MG: The establishment of a normal human population dose-response curve for lymphocytes cultured with PHA (phytohemagglutinin). Clin Exp Immunol 8:421-425, 1971. 19. Spitler LE, et al: The Wiskott-Aldrich syndrome: Results of transfer factor therapy. J Clin Invest 51:3216-3224, 1972.Crossref 20. Marsman AJW, et al: Migration inhibition experiments with mixtures of human peripheral lymphocytes and guinea pig peritoneal exudate. Europ J Immunol 2:546-550, 1972.Crossref 21. Wybran J, Chantler S, Fudenberg HN: Isolation of normal T cells in chronic lymphatic leukaemia. Lancet 1:126-129, 1973.Crossref 22. Vasquez AM, Kenny FM: Ovarian failure and antiovarian antibodies in association with hypoparathyroidism, moniliasis and Addison's and Hashimoto's diseases. Obstet Gynecol 41:414-418, 1973. 23. Golonka JE, Goodman AD: Coexistence of primary ovarian insufficiency, primary adrenocortical insufficiency and idiopathic hypoparathyroidism. J Clin Endocrinol Metab 28:79-82, 1968.Crossref 24. Irvine WJ, et al: Immunological aspects of premature ovarian failure associated with idiopathic Addison's disease. Lancet 2:883-887, 1968.Crossref 25. Kolb FO, et al: Primary hypoparathyroidism, Addison's disease and ovarian failure , in Irvine WJ, Loraine JA (eds): Reproductive Endocrinology . Edinburgh, E & S Livingstone Ltd, 1970, pp 116-121. 26. Drury MI, et al: Juvenile familial endocrinopathy. Clin Exp Immunol 7:125-132, 1970. 27. Block MB, et al: Immunological findings in familial juvenile endocrine deficiency syndrome associated with mucocutaneous candidiasis. Am J Med Sci 261:213-218, 1971.Crossref 28. Stankler L, Bewsher PD: Chronic mucocutaneous candidiasis, endocrine deficiency and alopecia areata. Br J Dermatol 86:238-245, 1972.Crossref 29. Sjoberg KH: Moniliasis—an internal disease? Three cases of idiopathic hypoparathyroidism with moniliasis, steatorrhea, primary amenorrhea and pernicious anemia. Acta Med Scand 179:157-166, 1966.Crossref 30. Spinner MW, Blizzard RM, Childs B: Clinical and genetic heterogeneity in idiopathic Addison's disease and hypoparathyroidism. J Clin Endocrinol Metab 28:795-804, 1968.Crossref 31. Steinberg H, Waldron BR: Idiopathic hypoparathyroidism: An analysis of 52 cases, including the report of a new case. Medicine 31:133-154,1952.Crossref 32. Forbes GB: Clinical features of idiopathic hypoparathyroidism in children. Ann NY Acad Sci 64:432-455, 1956-1957.Crossref 33. Kopin IJ, Rosenberg IN: Idiopathic hypoparathyroidism: Report of a case with autopsy findings. Ann Intern Med 53:1238-1249, 1960.Crossref 34. Turkington RW, Lebovitz HE: Extra-adrenal endocrine deficiencies in Addison's disease. Am J Med 43:499-507, 1967.Crossref 35. Strickland RG: Pernicious anemia and polyendocrine deficiency. Ann Intern Med 70:1001-1005, 1969.Crossref 36. Chilgren RA, et al: The cellular immune defect in chronic mucocutaneous candidiasis. Lancet 1:1286-1288, 1969.Crossref 37. Kirkpatrick CH: Chronic mucocutaneous candidiasis: Model-building in cellular immunity. Ann Intern Med 74:955-978, 1971.Crossref 38. Wells RS, et al: Familial chronic muco-cutaneous candidiasis. J Med Genet 9:302-310, 1972.Crossref 39. Gajl-Peczalska KJ, et al: B lymphocytes in Di-George syndrome. Lancet 1:1344-1345, 1972.Crossref 40. Kretschmer R, et al: Congenital aplasia of the thymus gland (DiGeorge's Syndrome). N Engl J Med 279:1295-1301, 1968.Crossref 41. Bankhurst AD, Torrigiani G, Allison AC: Lymphocytes binding human thyroglobulin in healthy people and its relevance to tolerance for autoantigens. Lancet 1:226-230, 1973.Crossref 42. Irvine WJ, Stewart AG, Scarth L: A clinical and immunological study of adreno-cortical insufficiency (Addison's disease). Clin Exp Immunol 2:31-69, 1967. 43. 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Archives of Internal Medicine – American Medical Association
Published: Nov 1, 1974
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