Department of Pediatrics, Takamatsu Red Cross Hospital, Takamatsu, Kagawa, Japan.
Department of Total Medical Support Center, Ehime University Hospital, Toon, Ehime, Japan.
PLoS One. 2021 Oct 1;16(10):e0258090. doi: 10.1371/journal.pone.0258090. eCollection 2021.
IgA nephropathy (IgAN) is the most common form of glomerulonephritis worldwide. Pediatric patients in Japan are diagnosed with IgAN at an early stage of the disease through annual urinary examinations. Tumor necrosis factor-like weak inducer of apoptosis (TWEAK) and fibroblast growth factor-inducible 14 (Fn14) have various roles, including proinflammatory effects, and modulation of several kidney diseases; however, no reports have described their roles in pediatric IgAN. In this study, we performed pathological and immunohistochemical analyses of samples from 14 pediatric IgAN patients. Additionally, gene expression arrays of glomeruli by laser-captured microdissection were performed in hemi-nephrectomized high serum IgA (HIGA) mice, a model of IgA nephropathy, to determine the role of Fn14. Glomeruli with intense Fn14 deposition were observed in 80% of mild IgAN cases; however, most severe cases showed glomeruli with little or no Fn14 deposition. Fn14 deposition was not observed in obvious mesangial proliferation or the crescent region of glomeruli, but was detected strongly in the glomerular tuft, with an intact appearance. In HIGA mice, Fn14 deposition was observed mildly beginning at 11 weeks of age, and stronger Fn14 deposition was detected at 14 weeks of age. Expression array analysis indicated that Fn14 expression was higher in HIGA mice at 6 weeks of age, increased slightly at 11 weeks, and then decreased at 26 weeks when compared with controls at equivalent ages. These findings suggest that Fn14 signaling affects early lesions but not advanced lesions in patients with IgAN. Further study of the TWEAK/Fn14 pathway will contribute to our understanding of the progression of IgAN.
IgA 肾病(IgAN)是全球最常见的肾小球肾炎类型。日本的儿科患者通过每年的尿液检查在疾病的早期阶段被诊断出患有 IgAN。肿瘤坏死因子样凋亡弱诱导物(TWEAK)和成纤维细胞生长因子诱导 14(Fn14)具有多种作用,包括促炎作用和调节几种肾脏疾病;然而,目前尚无报告描述它们在儿科 IgAN 中的作用。在这项研究中,我们对 14 名儿科 IgAN 患者的样本进行了病理和免疫组织化学分析。此外,还对半肾切除高血清 IgA(HIGA)小鼠(IgA 肾病模型)的肾小球进行了激光捕获微切割的基因表达阵列分析,以确定 Fn14 的作用。在 80%的轻度 IgAN 病例中观察到强烈的 Fn14 沉积的肾小球;然而,大多数严重病例显示肾小球的 Fn14 沉积很少或没有。在明显的系膜增生或肾小球的新月体区域未观察到 Fn14 沉积,但在肾小球簇中强烈检测到 Fn14 沉积,外观完整。在 HIGA 小鼠中,从 11 周龄开始观察到轻度的 Fn14 沉积,在 14 周龄时检测到更强的 Fn14 沉积。表达谱分析表明,HIGA 小鼠在 6 周龄时 Fn14 表达较高,在 11 周龄时略有增加,然后在 26 周龄时与同龄对照相比降低。这些发现表明,Fn14 信号在 IgAN 患者的早期病变中起作用,但在晚期病变中不起作用。对 TWEAK/Fn14 途径的进一步研究将有助于我们了解 IgAN 的进展。