Lymphocytic adenohypophysitis presenting as infertility

Case report

View More View Less
  • 1 Clinical Neurosurgery Section, Surgical Neurology Branch, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, Maryland
Restricted access

Purchase Now

USD  $45.00

JNS + Pediatrics - 1 year subscription bundle (Individuals Only)

USD  $505.00

JNS + Pediatrics + Spine - 1 year subscription bundle (Individuals Only)

USD  $600.00
Print or Print + Online

✓ The authors report a nulliparous patient presenting with infertility and hyperprolactinemia. She underwent transsphenoidal surgery after radiological investigation disclosed an enlarged pituitary gland which did not respond to bromocriptine therapy. The removed tissue had histological features consistent with adenohypophysitis including a diffuse lymphocytic infiltrate. The lymphocyte subsets present in the infiltrate were characterized by immunohistochemical methods to establish the contribution of different elements of the cellular immune response.

Lymphocytes bearing CD4 antigen (helper-inducer cells) were most prominent and appeared to bear the majority of the interleukin-2 receptor (expressed during lymphocytic activation) present in the pituitary gland. A few B lymphocytes were also observed. The location of the major histocompatibility antigen (classes I and II) and interleukin-2 receptor correlated with the lymphocytes and macrophages rather than with the stromal or parenchymal elements of the pituitary.

Lymphocytic adenohypophysitis is an unusual cause of pituitary enlargement which can mimic a pituitary tumor, and is sometimes associated with hyperprolactinemia. In women of child-bearing age, it almost always occurs during pregnancy or the postpartum stage. The autoimmune disorder reported here has not previously been associated with infertility nor has the lymphocytic infiltrate of the pituitary previously been analyzed in detail by modern immunological methods.

JNS + Pediatrics - 1 year subscription bundle (Individuals Only)

USD  $505.00

JNS + Pediatrics + Spine - 1 year subscription bundle (Individuals Only)

USD  $600.00

Contributor Notes

Address for Dr. McCutcheon: Montreal Neurological Institute, 3801 University Street, Montreal, Quebec, Canada.Address reprint requests to: Edward H. Oldfield, M.D., 9000 Rockville Pike, National Institutes of Health, Building 10, Room 5D37, Bethesda, Maryland 20892.
  • 1.

    Asa SL, , Bilbao JM, & Kovaks K, et al: Lymphocytic hypophysitis of pregnancy resulting in hypopituitarism: a distinct clinicopathologic entity. Ann Intern Med 95:166171, 1981 Asa SL, Bilbao JM, Kovaks K, et al: Lymphocytic hypophysitis of pregnancy resulting in hypopituitarism: a distinct clinicopathologic entity. Ann Intern Med 95:166–171, 1981

    • Search Google Scholar
    • Export Citation
  • 2.

    Baskin DS, , Townsend JJ, & Wilson CB: Lymphocytic adenohypophysitis of pregnancy simulating a pituitary adenoma: a distinct pathological entity. Report of two cases. J Neurosurg 56:148153, 1982 Baskin DS, Townsend JJ, Wilson CB: Lymphocytic adenohypophysitis of pregnancy simulating a pituitary adenoma: a distinct pathological entity. Report of two cases. J Neurosurg 56:148–153, 1982

    • Search Google Scholar
    • Export Citation
  • 3.

    Bottazzo G, , Dean BM, & McNally JM, et al: In situ characterization of autoimmune phenomena and expression of HLA molecules in the pancreas in diabetic insulitis. N Engl J Med 313:353360, 1985 Bottazzo G, Dean BM, McNally JM, et al: In situ characterization of autoimmune phenomena and expression of HLA molecules in the pancreas in diabetic insulitis. N Engl J Med 313:353–360, 1985

    • Search Google Scholar
    • Export Citation
  • 4.

    Bottazzo G, , Florin-Christiansen A, & Pouplard A, et al: Autoantibodies of prolactin-secreting cells of human pituitary. Lancet 2:97101, 1975 Bottazzo G, Florin-Christiansen A, Pouplard A, et al: Autoantibodies of prolactin-secreting cells of human pituitary. Lancet 2:97–101, 1975

    • Search Google Scholar
    • Export Citation
  • 5.

    Bottazzo G, , Pujol-Borrell R, & Hanafusa T, et al: Role of aberrant HLA-DR expression and antigen presentation in the induction of endocrine autoimmunity. Lancet 2:11151119, 1983 Bottazzo G, Pujol-Borrell R, Hanafusa T, et al: Role of aberrant HLA-DR expression and antigen presentation in the induction of endocrine autoimmunity. Lancet 2:1115–1119, 1983

    • Search Google Scholar
    • Export Citation
  • 6.

    Cebelin MS, , Velasco ME, & de las Mulas JM, et al: Galactorrhea associated with lymphocytic adenohypophysitis: case report. Br J Obstet Gynaecol 88:675680, 1981 Cebelin MS, Velasco ME, de las Mulas JM, et al: Galactorrhea associated with lymphocytic adenohypophysitis: case report. Br J Obstet Gynaecol 88:675–680, 1981

    • Search Google Scholar
    • Export Citation
  • 7.

    Cosman F, , Post KD, & Holub DA: Lymphocytic hypophysitis: report of 3 cases and review of the literature. Medicine 68:240256, 1989 Cosman F, Post KD, Holub DA: Lymphocytic hypophysitis: report of 3 cases and review of the literature. Medicine 68:240–256, 1989

    • Search Google Scholar
    • Export Citation
  • 8.

    Daar AS, , Fuggle SV, & Fabre JW, et al: The detailed distribution of MHC class II antigens in normal human organs. Transplantation 38:293298, 1984 Daar AS, Fuggle SV, Fabre JW, et al: The detailed distribution of MHC class II antigens in normal human organs. Transplantation 38:293–298, 1984

    • Search Google Scholar
    • Export Citation
  • 9.

    Del Prete GF, , Maggi E, & Mariotti S, et al: Cytolytic T lymphocytes with natural killer activity in thyroid infiltrate of patients with Hashimoto's thyroiditis: analysis at the clonal level. J Clin Endocrinol Metab 62:16, 1986 Del Prete GF, Maggi E, Mariotti S, et al: Cytolytic T lymphocytes with natural killer activity in thyroid infiltrate of patients with Hashimoto's thyroiditis: analysis at the clonal level. J Clin Endocrinol Metab 62:1–6, 1986

    • Search Google Scholar
    • Export Citation
  • 10.

    Engelberth O, & Jeová Z: Autoantibodies in Sheehan's syndrome. Lancet 1:1075, 1965 (Letter) Engelberth O, Jeová Z: Autoantibodies in Sheehan's syndrome. Lancet 1:1075, 1965 (Letter)

    • Search Google Scholar
    • Export Citation
  • 11.

    Goudie RB, & Pinkerton PH: Anterior hypophysitis and Hashimoto's disease in a young woman. J Pathol Bacterid 83:584585, 1962 Goudie RB, Pinkerton PH: Anterior hypophysitis and Hashimoto's disease in a young woman. J Pathol Bacterid 83:584–585, 1962

    • Search Google Scholar
    • Export Citation
  • 12.

    Guay AT, , Agnello V, & Tronic BC, et al: Lymphocytic hypophysitis in a man. J Clin Endocrinol Metab 64:631634, 1987 Guay AT, Agnello V, Tronic BC, et al: Lymphocytic hypophysitis in a man. J Clin Endocrinol Metab 64:631–634, 1987

    • Search Google Scholar
    • Export Citation
  • 13.

    Hsu SM, , Raine L, & Fanger H: Use of avidin-biotin-peroxidase complex (ABC) in immunoperoxidase techniques: a comparison between ABC and unlabeled antibody (PAP) procedures. J Histochem Cytochem 29:577580, 1981 Hsu SM, Raine L, Fanger H: Use of avidin-biotin-peroxidase complex (ABC) in immunoperoxidase techniques: a comparison between ABC and unlabeled antibody (PAP) procedures. J Histochem Cytochem 29:577–580, 1981

    • Search Google Scholar
    • Export Citation
  • 14.

    Hume R, & Roberts GH: Hypophysitis and hypopituitarism: report of a case. Br Med J 2:548550, 1967 Hume R, Roberts GH: Hypophysitis and hypopituitarism: report of a case. Br Med J 2:548–550, 1967

    • Search Google Scholar
    • Export Citation
  • 15.

    Jansson R, , Karlsson A, & Forsum U: Intrathyroid HLA-DR expression and nodular colloid goitre. Clin Exp Immunol 58:264272, 1985 Jansson R, Karlsson A, Forsum U: Intrathyroid HLA-DR expression and nodular colloid goitre. Clin Exp Immunol 58:264–272, 1985

    • Search Google Scholar
    • Export Citation
  • 16.

    Jensen MD, , Handwerger BS, & Scheithauer BW, et al: Lymphocytic hypophysitis with isolated corticotropin deficiency. Ann Intern Med 105:200203, 1986 Jensen MD, Handwerger BS, Scheithauer BW, et al: Lymphocytic hypophysitis with isolated corticotropin deficiency. Ann Intern Med 105:200–203, 1986

    • Search Google Scholar
    • Export Citation
  • 17.

    Klein I, , Draus KE, & Martinez AJ, et al: Evidence for cellular mediated immunity in an animal model of autoimmune pituitary disease. Endocrinol Res Commun 9:145153, 1982 Klein I, Draus KE, Martinez AJ, et al: Evidence for cellular mediated immunity in an animal model of autoimmune pituitary disease. Endocrinol Res Commun 9:145–153, 1982

    • Search Google Scholar
    • Export Citation
  • 18.

    Kroczek RA, , Black CDV, & Barbet J, et al: Induction of IL-2 receptor expression in vivo: response to allogeneic cells. Transplantation 44:547553, 1987 Kroczek RA, Black CDV, Barbet J, et al: Induction of IL-2 receptor expression in vivo: response to allogeneic cells. Transplantation 44:547–553, 1987

    • Search Google Scholar
    • Export Citation
  • 19.

    Laws ER Jr, , Fode NC, & Randall RV, et al: Pregnancy following transsphenoidal resection of prolactin-secreting pituitary tumors. J Neurosurg 58:685688, 1983 Laws ER Jr, Fode NC, Randall RV, et al: Pregnancy following transsphenoidal resection of prolactin-secreting pituitary tumors. J Neurosurg 58:685–688, 1983

    • Search Google Scholar
    • Export Citation
  • 20.

    Levine S: Allergic adenohypophysitis: new experimental disease of the pituitary gland. Science 158:11901191, 1967 Levine S: Allergic adenohypophysitis: new experimental disease of the pituitary gland. Science 158:1190–1191, 1967

    • Search Google Scholar
    • Export Citation
  • 21.

    Lindhal G, , Hedfors E, & Klareskog L: Epithelial HLA-DR expression and T lymphocyte subsets in salivary glands in Sjögren's syndrome. Clin Exp Immunol 61:475482, 1985 Lindhal G, Hedfors E, Klareskog L: Epithelial HLA-DR expression and T lymphocyte subsets in salivary glands in Sjögren's syndrome. Clin Exp Immunol 61:475–482, 1985

    • Search Google Scholar
    • Export Citation
  • 22.

    Lord RHH, , Hancock WW, & Colby AJ, et al: Effects of anti-IL 2 receptor monoclonal antibody and cyclosporine on IL 2 receptor-positive cells infiltrating cardiac allografts in the rat. Transplant Proc 19:354355, 1987 Lord RHH, Hancock WW, Colby AJ, et al: Effects of anti-IL 2 receptor monoclonal antibody and cyclosporine on IL 2 receptor-positive cells infiltrating cardiac allografts in the rat. Transplant Proc 19:354–355, 1987

    • Search Google Scholar
    • Export Citation
  • 23.

    Ludwig H, & Schernthaner G: Multiorganspezifische Autoimmunität bei Idiopatischer Nebenniereninsuffizienz. Wien Klin Wochenschr 90:736741, 1978 Ludwig H, Schernthaner G: Multiorganspezifische Autoimmunität bei Idiopatischer Nebenniereninsuffizienz. Wien Klin Wochenschr 90:736–741, 1978

    • Search Google Scholar
    • Export Citation
  • 24.

    Madsen FF, , Døhr O, & Ludwigsen E: Lymphocytic adenohypophysitis: a review and a case. Danish Med Bull 32:185188, 1985 Madsen FF, Døhr O, Ludwigsen E: Lymphocytic adenohypophysitis: a review and a case. Danish Med Bull 32:185–188, 1985

    • Search Google Scholar
    • Export Citation
  • 25.

    Mayfield RK, , Levine JH, & Gordon L, et al: Lymphoid adenohypophysitis presenting as a pituitary tumor. Am J Med 69:619623, 1980 Mayfield RK, Levine JH, Gordon L, et al: Lymphoid adenohypophysitis presenting as a pituitary tumor. Am J Med 69:619–623, 1980

    • Search Google Scholar
    • Export Citation
  • 26.

    Mazzone P, , Kelly W, & Ensinck J: Lymphocytic hypophysitis associated with antiparietal cell antibodies and vitamin B12 deficiency. Arch Intern Med 143:17941795, 1983 Mazzone P, Kelly W, Ensinck J: Lymphocytic hypophysitis associated with antiparietal cell antibodies and vitamin B12 deficiency. Arch Intern Med 143:1794–1795, 1983

    • Search Google Scholar
    • Export Citation
  • 27.

    McDermott MW, , Griesdale DE, & Berry K, et al: Lymphocytic adenohypophysitis. Can J Neurol Sci 15:3843, 1988 McDermott MW, Griesdale DE, Berry K, et al: Lymphocytic adenohypophysitis. Can J Neurol Sci 15:38–43, 1988

    • Search Google Scholar
    • Export Citation
  • 28.

    McGrail KM, , Beyerl BD, & Black PM: Lymphocytic adenohypophysitis of pregnancy with complete recovery. Neurosurgery 20:791793, 1987 McGrail KM, Beyerl BD, Black PM: Lymphocytic adenohypophysitis of pregnancy with complete recovery. Neurosurgery 20:791–793, 1987

    • Search Google Scholar
    • Export Citation
  • 29.

    Miura M, , Ushio Y, & Kuratsu J, et al: Lymphocytic adenohypophysitis: report of two cases. Surg Neurol 32:463470, 1989 Miura M, Ushio Y, Kuratsu J, et al: Lymphocytic adenohypophysitis: report of two cases. Surg Neurol 32:463–470, 1989

    • Search Google Scholar
    • Export Citation
  • 30.

    Portocarrero CJ, , Robinson AG, & Taylor AL, et al: Lymphoid hypophysitis: an unusual cause of hyperprolactinemia and enlarged sella turcica. JAMA 246:18111812, 1981 Portocarrero CJ, Robinson AG, Taylor AL, et al: Lymphoid hypophysitis: an unusual cause of hyperprolactinemia and enlarged sella turcica. JAMA 246:1811–1812, 1981

    • Search Google Scholar
    • Export Citation
  • 31.

    Quencer RM: Lymphocytic adenohypophysitis: autoimmune disorder of the pituitary gland. AJNR 1:343345, 1980 Quencer RM: Lymphocytic adenohypophysitis: autoimmune disorder of the pituitary gland. AJNR 1:343–345, 1980

    • Search Google Scholar
    • Export Citation
  • 32.

    Richtsmeier AJ, , Henry RA, & Bloodworth JMB Jr, et al: Lymphoid hypophysitis with selective adrenocorticotropic hormone deficiency. Arch Intern Med 140:12431245, 1980 Richtsmeier AJ, Henry RA, Bloodworth JMB Jr, et al: Lymphoid hypophysitis with selective adrenocorticotropic hormone deficiency. Arch Intern Med 140:1243–1245, 1980

    • Search Google Scholar
    • Export Citation
  • 33.

    Shapiro ME, , Kirkman RL, & Reed MH, et al: Monoclonal anti-IL-2 receptor antibody in primary renal transplantation. Transplant Proc 19:594598, 1987 Shapiro ME, Kirkman RL, Reed MH, et al: Monoclonal anti-IL-2 receptor antibody in primary renal transplantation. Transplant Proc 19:594–598, 1987

    • Search Google Scholar
    • Export Citation
  • 34.

    Sobrinho-Simoes M, , Brandao A, & Paiva ME, et al: Lymphoid hypophysitis in a patient with lymphoid thyroiditis, lymphoid adrenalitis and idiopathic retroperitoneal fibrosis. Arch Pathol Lab Med 109:230233, 1985 Sobrinho-Simoes M, Brandao A, Paiva ME, et al: Lymphoid hypophysitis in a patient with lymphoid thyroiditis, lymphoid adrenalitis and idiopathic retroperitoneal fibrosis. Arch Pathol Lab Med 109:230–233, 1985

    • Search Google Scholar
    • Export Citation
  • 35.

    Speroff L, , Glass RH, & Kase NG: Clinical Gynecologic Endocrinology and Infertility. Baltimore: Williams & Wilkins, 1989, p 214 Speroff L, Glass RH, Kase NG: Clinical Gynecologic Endocrinology and Infertility. Baltimore: Williams & Wilkins, 1989, p 214

    • Search Google Scholar
    • Export Citation
  • 36.

    Wild RA, & Kepley M: Lymphocytic hypophysitis in a patient with amenorrhea and hyperprolactinemia: a case report. J Reprod Med 31:211216, 1986 Wild RA, Kepley M: Lymphocytic hypophysitis in a patient with amenorrhea and hyperprolactinemia: a case report. J Reprod Med 31:211–216, 1986

    • Search Google Scholar
    • Export Citation

Metrics

All Time Past Year Past 30 Days
Abstract Views 514 179 4
Full Text Views 167 5 0
PDF Downloads 92 6 3
EPUB Downloads 0 0 0