Division of Anatomic Pathology, Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, USA.
Division of Nephrology and Hypertension, Mayo Clinic, Rochester, MN, USA.
Pediatr Nephrol. 2017 Sep;32(9):1625-1633. doi: 10.1007/s00467-017-3702-8. Epub 2017 Jun 7.
C3 glomerulopathy (C3G) is rare type of glomerulonephritis resulting from the glomerular deposition of C3 due to dysregulation of the alternative pathway of complement. It is further subdivided into C3 glomerulonephritis (C3GN) and dense deposit disease (DDD), depending on the ultrastructural features. C3GN usually presents with a membranoproliferative pattern of injury. Crescents may or may not be present. However, we have noted a severe necrotizing and crescentic glomerulonephritis in a small subset of C3GN patients.
CASE DIAGNOSIS/TREATMENT: We present the clinical features, kidney biopsy findings, complement evaluation, treatment, and follow-up of four C3GN patients. We also compare the findings with four DDD patients with a severe necrotizing and crescentic glomerulonephritis. The patients were young and presented with hematuria and proteinuria. The biopsy was remarkable for the large number of crescents, in association with bright glomerular staining for C3. The patients were treated with steroids/immunosuppressive drugs. Three of the 4 C3GN patients and 2 of the 4 DDD patients showed stable renal function at follow-up. We find remarkable similarities between our C3GN patients with crescents and the previously described entity of juvenile acute nonproliferative glomerulonephritis.
To summarize, C3G with a severe crescentic phenotype is rare, affects children and young adults, and has a variable response to steroid and immunosuppressive treatment. It is important to recognize this rare cause of crescentic glomerulonephritis so that appropriate evaluation and treatment can be carried out.
C3 肾小球病(C3G)是一种罕见的肾小球肾炎,由于补体替代途径的失调导致 C3 在肾小球中沉积。它进一步细分为 C3 肾小球肾炎(C3GN)和致密沉积物病(DDD),这取决于超微结构特征。C3GN 通常表现为膜增生性损伤模式。新月体可能存在也可能不存在。然而,我们在一小部分 C3GN 患者中注意到一种严重的坏死性和新月体性肾小球肾炎。
病例诊断/治疗:我们介绍了 4 例 C3GN 患者的临床特征、肾活检结果、补体评估、治疗和随访情况,并将这些发现与 4 例伴有严重坏死性和新月体性肾小球肾炎的 DDD 患者进行了比较。患者年龄较小,表现为血尿和蛋白尿。活检的特征是大量新月体,同时 C3 在肾小球中呈明亮染色。患者接受了类固醇/免疫抑制剂治疗。4 例 C3GN 患者中的 3 例和 4 例 DDD 患者中的 2 例在随访时肾功能稳定。我们发现我们的 C3GN 患者中有新月体的患者与以前描述的青少年急性非增殖性肾小球肾炎之间存在显著相似性。
综上所述,伴有严重新月体表型的 C3G 较为罕见,影响儿童和年轻人,对类固醇和免疫抑制剂治疗的反应不一。重要的是要认识到这种罕见的新月体性肾小球肾炎的病因,以便进行适当的评估和治疗。