Antipituitary antibodies against gonadotropin-secreting cells in adult male patients with apparently idiopathic hypogonadotropic hypogonadism
Antipituitary antibodies against gonadotropin-secreting cells in adult male patients with apparently idiopathic hypogonadotropic hypogonadism
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DOI：
10.1210/jc.2006-1216
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发表时间：
2007-02-01
影响因子：
5.8
通讯作者：
Bellastella, Antonio
中科院分区：
文献类型：
--
作者：
De Bellis, Annamaria;Sinisi, Antonio Agostino;Bellastella, Antonio
内容：低促性腺激素性性腺功能减退症（HH）可以发生在生命的任何阶段，作为一个孤立的先天性或获得性异常或在更广泛的垂体或下丘脑损害。然而，在特发性HH患者的缺陷仍然是未知的。目的：本研究的目的是调查一组HH患者的抗垂体抗体（阿帕）的患病率与或不卡尔曼综合征和表征他们的垂体靶。设计：我们进行了一项横断面队列研究。设置：这项研究是在那不勒斯第二大学内分泌科进行。患者：21例嗅觉正常的HH患者（第1组），10例Kallmann综合征患者（第2组），13例HH伴其他垂体激素缺乏症患者（第3组）和50名正常对照者进行了研究。此外，磁共振成像（MRI）的下丘脑-垂体区进行了所有三组patients.Results：阿帕检测在高滴度的8个21例在第1组（38%）和5个13在第3组（38.4%），并在低滴度的2个10在第2组（20%）和3个50对照组（6%）。在第1组患者中，阿帕免疫染色选择性促性腺激素分泌细胞，而在第3组中，他们也免疫染色其他垂体腺瘤分泌细胞。第1组中没有患者在MRI上显示改变，而第2组中的所有患者均显示嗅球/嗅束和/或嗅沟发育不全/发育不全。在第3组中的5个APA阳性患者中，有三个正常的MRI，空鞍的结果，和一个有结果的自身免疫性hypophysiitis.Conclusions：我们的研究结果表明，一些明显的特发性HH的情况下，孤立的和与其他垂体损伤，可以引起的早期自身免疫过程中涉及促性腺激素在垂体水平。未来的纵向研究需要澄清这一过程的自然史和早期皮质类固醇治疗的可能影响。
Context: Hypogonadotropic hypogonadism (HH) can occur at any stage of life as an isolated congenital or acquired abnormality or within a more generalized pituitary or hypothalamic impairment. However, the defect in patients with idiopathic HH is still unknown.Objective: The aim of this study was to investigate the prevalence of antipituitary antibodies (APA) in a group of HH patients with or without Kallmann's syndrome and to characterize their pituitary target.Design: We conducted a cross-sectional cohort study.Setting: The study was performed at the Endocrinology Unit of the Second University of Naples.Patients: Twenty-one HH patients with normal sense of smell (group 1), 10 patients with Kallmann's syndrome (group 2), 13 patients with HH associated with other pituitary hormone deficiencies (group 3), and 50 normal controls were studied.Main Outcome Measures: APA were evaluated in patients and in controls by indirect immunofluorescence. Moreover, a magnetic resonance imaging (MRI) of the hypothalamic-pituitary region was performed in all three groups of patients.Results: APA were detected at high titer in eight out of 21 patients in group 1 (38%) and in five of 13 in group 3 (38.4%), and at low titers in two out of 10 in group 2 (20%) and in three of 50 controls (6%). In patients of group 1, APA immunostained selectively gonadotropin-secreting cells, whereas in those of group 3, they immunostained other pituitary hormone-secreting cells also. None of patients in group 1 showed alterations on MRI, whereas all patients in group 2 showed aplasia/hypoplasia of the olfactory bulbs/tracts and/or of olfactory sulci. Among the five APA-positive patients in group 3, three had normal MRI, one had findings of empty sella, and one had findings of autoimmune hypophysitis.Conclusions: Our results suggest that some apparently idiopathic cases of HH, both isolated and associated with other pituitary impairment, can be caused by an early autoimmune process involving the gonadotrophs at pituitary level. Future longitudinal studies are needed to clarify the natural history of this process and the possible effect of early corticosteroid therapy.