Literature DB >> 14557429

The hypoparathyroidism of autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy protective effect of male sex.

Mikhail Gylling1, Essi Kääriäinen, Riitta Väisänen, Laura Kerosuo, Marja-Liisa Solin, Leena Halme, Seppo Saari, Maria Halonen, Olle Kämpe, Jaakko Perheentupa, Aaro Miettinen.   

Abstract

In autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy, hypoparathyroidism (HP) is the most common endocrine component. It occurs in most (but not all) patients. Determinants of its occurrence are unknown, and there is no proof for its autoimmune nature. Recently, the Ca(2+)-sensing receptor (CaSR) was reported to be an autoantigen in HP. With our group of 90 patients, we aimed at identifying the determinants and pathomechanism of HP. For the determinants, we evaluated gender and the HLA class II. For the pathomechanism, we searched for parathyroid autoantibodies, including antibodies against CaSR and PTH. Also, we studied whether AIRE is expressed in the human parathyroid, because its absence could be a pathogenetic factor. We found a clear gender linkage with lower and later incidence in males. Of the 14 patients who had escaped HP, 13 were males. This was associated with adrenal failure, which was the first or only endocrinopathy in 47% of males vs. 7% of females. In contrast, we found no linkage to the HLA class II. By immunofluorescence, 19% of the patients had antibodies to parathyroid epithelia. By immunoblotting, these recognized several parathyroid proteins. No antibodies were observed against the CaSR or PTH. By RT-PCR, AIRE mRNA was not found in the parathyroid.

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Year:  2003        PMID: 14557429     DOI: 10.1210/jc.2003-030700

Source DB:  PubMed          Journal:  J Clin Endocrinol Metab        ISSN: 0021-972X            Impact factor:   5.958


  7 in total

Review 1.  Autoimmune polyendocrinopathy candidiasis ectodermal dystrophy (APECED): a model disease to study molecular aspects of endocrine autoimmunity.

Authors:  P Peterson; J Pitkänen; N Sillanpää; K Krohn
Journal:  Clin Exp Immunol       Date:  2004-03       Impact factor: 4.330

2.  Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy.

Authors:  Choudhary Sonal; McLeod Michael; Torchia Daniele; Romanelli Paolo
Journal:  J Clin Aesthet Dermatol       Date:  2012-12

Review 3.  Autoimmune polyendocrinopathy candidiasis ectodermal dystrophy.

Authors:  Kai Kisand; Pärt Peterson
Journal:  J Clin Immunol       Date:  2015-07-05       Impact factor: 8.317

Review 4.  Clues to immune tolerance: the monogenic autoimmune syndromes.

Authors:  Michael Waterfield; Mark S Anderson
Journal:  Ann N Y Acad Sci       Date:  2010-10-22       Impact factor: 5.691

Review 5.  Immune deficiency disorders with autoimmunity and abnormalities in immune regulation-monogenic autoimmune diseases.

Authors:  Heather K Lehman; Mark Ballow
Journal:  Clin Rev Allergy Immunol       Date:  2008-04       Impact factor: 8.667

6.  New splice site acceptor mutation in AIRE gene in autoimmune polyendocrine syndrome type 1.

Authors:  Mireia Mora; Felicia A Hanzu; Marta Pradas-Juni; Gloria B Aranda; Irene Halperin; Manuel Puig-Domingo; Sira Aguiló; Eduardo Fernández-Rebollo
Journal:  PLoS One       Date:  2014-07-02       Impact factor: 3.240

7.  Autoimmune polyendocrinopathy candidiasis ectodermal dystrophy: insights into genotype-phenotype correlation.

Authors:  Donatella Capalbo; Lucia De Martino; Giuliana Giardino; Raffaella Di Mase; Iolanda Di Donato; Giancarlo Parenti; Pietro Vajro; Claudio Pignata; Mariacarolina Salerno
Journal:  Int J Endocrinol       Date:  2012-10-22       Impact factor: 3.257

  7 in total

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