DOI: http://dx.doi.org/10.18203/2320-6012.ijrms20161461

Autoimmune hypophysitis: a study of natural course

Vijaya Sarathi, Anish Kolly

Abstract


Background: Autoimmune hypophysitis is a rare autoimmune endocrinopathy. Literature on natural history of autoimmune hypophysitis is scarce.

Methods: We prospectively studied patients with autoimmune hypophysitis between January 2013 to June 2015 and all subjects were followed for at least 6 months. Autoimmune hypophysitis was diagnosed based on clinicoradiologic findings. All patients diagnosed with autoimmune hypophysitis were followed every three monthly with evaluation for pituitary functions and six monthly with gadolinium enhanced MRI of pituitary. Deficient hormones were replaced and none of the patients were treated with immunosuppressive therapy or surgery.

Results: Ten cases of autoimmune hypophysitis were diagnosed during the study period. Adrenocorticotropin deficiency was the most common (7/10) followed by thyroid stimulating hormone and gonadotropins (each in 5). Pituitary enlargement and stalk thickening were the most common imaging findings. Three patients had recovery of one hormone in each and none of the patients had progression of mass or new hormone deficiency on follow-up.

Conclusions: The study describes natural history of autoimmune hypophysitis in 10 patients from a single center. We suggest against the routine use of immunosuppressive therapy or surgery in patients with autoimmune hypophysitis.


Keywords


Autoimmune hypophysitis, Lymphocytic hypophysitis, Empty sella, Stalk thickening

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References


Tuğcu B, Gunaldi O, Postalci L, Tanriverdi O, Ofluoglu E, Sever N. Lymphocytic hypophysitis: an underestimated disease affecting the sellar region. Neurol Neurochir Pol. 2011;45(2):174-9.

Caturegli P. Autoimmune hypophysitis: an underestimated disease in search of its autoantigen(s). J Clin Endocrinol Metab. 2007;92(6):2038-40.

Caturegli P, Lupi I, Landek-Salgado M, Kimura H, Rose NR. Pituitary autoimmunity: 30 years later. Autoimmun Rev. 2008;7(8):631-7.

Khare S, Jagtap VS, Budyal SR, Kasaliwal R, Kakade HR, Bukan A, et al. Primary (autoimmune) hypophysitis: a single centre experience. Pituitary. 2015;18(1):16-22.

Lammert A, Schneider HJ, Bergmann T, Benck U, Krämer BK, Gärtner R, et al. Hypophysitis caused by ipilimumab in cancer patients: hormone replacement or immunosuppressive therapy. Exp Clin Endocrinol Diabetes. 2013;121(10):581-7.

Caturegli P, Newschaffer C, Olivi A, Pomper MG, Burger PC, Rose NR. Autoimmune hypophysitis. Endocr Rev. 2005;26(5):599-614.

Honegger J, Schlaffer S, Menzel C, Droste M, Werner S, Elbelt U, et al. Pituitary Working Group of the German Society of Endocrinology. Diagnosis of Primary Hypophysitis in Germany. J Clin Endocrinol Metab. 2015;100(10):3841-9.

Faje AT, Sullivan R, Lawrence D, Tritos NA, Fadden R, Klibanski A, et al. Ipilimumab-induced hypophysitis: a detailed longitudinal analysis in a large cohort of patients with metastatic melanoma. J Clin Endocrinol Metab. 2014;99(11):4078-85.

Bashier AM, Harifi G, Abdelgadir EI. Igg4-Related Disorder and Endocrine Diseases: A Review of the Literature. J Diabetes Metab. 2015;6:631.

Bottazzo GF, Pouplard A, Florin-Christensen A, Doniach D. Autoantibodies to prolactin-secreting cells of human pituitary. Lancet. 1975; 2(7925):97-101.

Crock PA, Salvi M, Miller A, Wall J, Guyda H. Detection of anti-pituitary antibodies by immunoblotting. J Immunol Methods. 1993;162:31-40.

Heaney AP, Sumerel B1, Rajalingam R, Bergsneider M, Yong WH, Liau LM. HLA Markers DQ8 and DR53 Are Associated With Lymphocytic Hypophysitis and May Aid in Differential Diagnosis. J Clin Endocrinol Metab. 2015;100(11):4092-7.

Bando H, Iguchi G, Fukuoka H, Taniguchi M, Yamamoto M, Matsumoto R, et al. The prevalence of IgG4-related hypophysitis in 170 consecutive patients with hypopituitarism and/or central diabetes insipidus and review of the literature. Eur J Endocrinol. 2013;170(2):161-72.

Honegger J, Buchfelder M, Schlaffer S, Droste M, Werner S, Strasburger C, et al; Pituitary Working Group of the German Society of Endocrinology. Treatment of Primary Hypophysitis in Germany. J Clin Endocrinol Metab. 2015;100(9):3460-9.

Mittal R, Kalra P, Dharmalingam M, Verma RG, Kulkarni S, Shetty P. Lymphocytic hypophysitis masquerading as pituitary adenoma. Indian J Endocrinol Metab. 2012;16(Suppl 2):S304-6.

Gutenberg A, Larsen J, Lupi I, Rohde V, Caturegli P. A radiologic score to distinguish autoimmune hypophysitis from nonsecreting pituitary adenoma preoperatively. AJNR Am J Neuroradiol. 2009;30:1766-72.

Hrubisková K, Jackuliak P, Vanuga P, Pura M, Payer J. Autoimmune polyendocrine syndrome type 2 associated with autoimmune hypophysitis and coeliac disease. Vnitr Lek. 2010;56:1169-76.