Gylling Mikhail, Kääriäinen Essi, Väisänen Riitta, Kerosuo Laura, Solin Marja-Liisa, Halme Leena, Saari Seppo, Halonen Maria, Kämpe Olle, Perheentupa Jaakko, Miettinen Aaro
Department of Bacteriology and Immunology, University of Helsinki, FIN-00014 Helsinki, Finland.
J Clin Endocrinol Metab. 2003 Oct;88(10):4602-8. doi: 10.1210/jc.2003-030700.
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.
在自身免疫性多内分泌腺病-念珠菌病-外胚层营养不良中,甲状旁腺功能减退症(HP)是最常见的内分泌组成部分。它在大多数(但并非所有)患者中出现。其发生的决定因素尚不清楚,也没有证据证明其自身免疫性质。最近,有报道称钙敏感受体(CaSR)是HP中的一种自身抗原。我们以90名患者组成的队列,旨在确定HP的决定因素和发病机制。对于决定因素,我们评估了性别和HLA II类。对于发病机制,我们寻找甲状旁腺自身抗体,包括针对CaSR和甲状旁腺激素(PTH)的抗体。此外,我们研究了自身免疫调节因子(AIRE)是否在人甲状旁腺中表达,因为其缺失可能是一个致病因素。我们发现了明显的性别关联,男性的发病率较低且出现较晚。在14名未发生HP的患者中,有13名是男性。这与肾上腺功能衰竭有关,肾上腺功能衰竭是47%的男性的首个或唯一内分泌病,而女性中这一比例为7%。相比之下,我们未发现与HLA II类的关联。通过免疫荧光法,19%的患者有针对甲状旁腺上皮的抗体。通过免疫印迹法,这些抗体识别了几种甲状旁腺蛋白。未观察到针对CaSR或PTH的抗体。通过逆转录聚合酶链反应(RT-PCR),在甲状旁腺中未发现AIRE信使核糖核酸(mRNA)。