Department of Pediatrics, Kobe University Graduate School of Medicine, 7-5-1 Kusunoki-cho, Chuo-ku, Kobe, Hyogo, 650-0017, Japan.
Department of Nephrology, Japanese Red Cross Nagoya Daini Hospital, 2-9 Myoken-cho, Showa-ku, Nagoya, Aichi, 466-8650, Japan.
Clin Exp Nephrol. 2021 Jul;25(7):779-787. doi: 10.1007/s10157-021-02054-3. Epub 2021 Mar 20.
Pathological findings in Alport syndrome frequently show mesangial proliferation and sometimes incidental IgA deposition, in addition to unique glomerular basement membrane (GBM) changes including thin basement membrane and/or lamellation. However, similar GBM abnormalities are also often observed in IgA nephropathy. Both diseases are also known to show hematuria, proteinuria, and sometimes macrohematuria when associated with viral infection. Therefore, it can be difficult to make a differential diagnosis, even based on clinical and pathological findings. Some recent articles demonstrated that galactose-deficient IgA1 (Gd-IgA1)-specific monoclonal antibody (KM55) could potentially enable incidental IgA deposition to be distinguished from IgA nephropathy.
We performed comprehensive gene screening and glomerular Gd-IgA1 and type IV collagen α5 chain immunostaining for five cases with both IgA deposition and GBM changes to confirm that Gd-IgA1 can help to distinguish these two diseases.
Four of the cases were genetically diagnosed with Alport syndrome (Cases 1-4) and one was IgA nephropathy with massive GBM changes, which had a negative gene test result (Case 5). In Cases 1-4, glomerular Gd-IgA1 deposition was not detected, although there was positivity for IgA in the mesangial area. In Case 5, glomerular Gd-IgA1 deposition was observed.
Gd-IgA1 expression analysis could clearly differentiate these two disorders. This approach can be applied to identify these two diseases showing identical clinical and pathological findings.
阿尔波特综合征的病理表现常伴有系膜增生,有时偶然伴有 IgA 沉积,此外还伴有独特的肾小球基底膜(GBM)改变,包括基底膜变薄和/或板层化。然而,IgA 肾病也常观察到类似的 GBM 异常。这两种疾病在与病毒感染相关时也会出现血尿、蛋白尿,有时还会出现肉眼血尿。因此,即使基于临床和病理发现,也很难做出鉴别诊断。一些最近的文章表明,半乳糖缺乏 IgA1(Gd-IgA1)特异性单克隆抗体(KM55)可能有助于将偶然的 IgA 沉积与 IgA 肾病区分开来。
我们对 5 例既有 IgA 沉积又有 GBM 改变的病例进行了全面的基因筛查和肾小球 Gd-IgA1 和 IV 型胶原α5 链免疫染色,以确认 Gd-IgA1 有助于区分这两种疾病。
4 例病例(病例 1-4)通过基因检测确诊为阿尔波特综合征,1 例(病例 5)为伴有大量 GBM 改变的 IgA 肾病,基因检测结果为阴性。在病例 1-4 中,虽然系膜区 IgA 呈阳性,但未检测到肾小球 Gd-IgA1 沉积。在病例 5 中,观察到肾小球 Gd-IgA1 沉积。
Gd-IgA1 表达分析可以明确区分这两种疾病。这种方法可用于识别具有相同临床和病理表现的这两种疾病。