Komatsuda Atsushi, Masai Rie, Ohtani Hiroshi, Togashi Masaru, Maki Nobuki, Sawada Ken-ichi, Wakui Hideki
Third Department of Internal Medicine, Akita University School of Medicine, Akita City, Akita 010-8543, Japan.
Nephrol Dial Transplant. 2008 Dec;23(12):3888-94. doi: 10.1093/ndt/gfn363. Epub 2008 Jul 2.
Very few cases of non-organized and non-Randall-type monoclonal immunoglobulin deposition disease (MIDD) associated with membranous features have been reported. Information on clinicopathological features and prognosis in this entity is limited.
We reviewed 5443 renal biopsies processed at our department, and identified three patients with MIDD associated with membranous features. We evaluated clinicopathological features and outcomes in these patients.
All patients had proteinuria, and one patient developed nephrotic syndrome. Renal insufficiency was not observed. Cryoglobulin or monoclonal protein in serum and urine was not detected. A renal biopsy showed thickening of the glomerular capillary walls and spike formation. Tubulointerstitial and vascular alterations were mild or absent. Immunofluorescence studies revealed granular IgG3-kappa deposits in two patients and IgG1-kappa deposits in one patient, along the glomerular capillary walls. Immunofluorescence studies using antibodies specific for gamma-heavy chain Fab containing C(H)1 domain, C(H)2 domain and C(H)3 domain did not show any apparent deletion. On confocal microscopy, glomerular colocalization of light and heavy chains was observed. Electron microscopy showed predominant subepithelial granular deposits without distinct ultrastructural organization. All patients were treated with steroids, and good effects were observed. A follow-up renal biopsy performed in one patient showed histological improvements. No patient developed myeloma or other haematological malignancy during the course of follow-up (mean 44 months).
MIDD associated with membranous features is an extremely rare but distinctive entity. Our study suggests glomerular deposition of a nondeleted whole immunoglobulin molecule. Patients with this entity appear to respond well to steroid therapy.
与膜性特征相关的非组织化且非兰德尔型单克隆免疫球蛋白沉积病(MIDD)的病例报道极少。关于该实体的临床病理特征和预后的信息有限。
我们回顾了在本部门处理的5443例肾活检病例,确定了3例具有膜性特征的MIDD患者。我们评估了这些患者的临床病理特征和结局。
所有患者均有蛋白尿,1例患者发展为肾病综合征。未观察到肾功能不全。未检测到血清和尿液中的冷球蛋白或单克隆蛋白。肾活检显示肾小球毛细血管壁增厚和钉突形成。肾小管间质和血管改变轻微或无改变。免疫荧光研究显示,2例患者沿肾小球毛细血管壁有颗粒状IgG3-κ沉积,1例患者有IgG1-κ沉积。使用针对包含C(H)1结构域、C(H)2结构域和C(H)3结构域的γ重链Fab的特异性抗体进行的免疫荧光研究未显示任何明显缺失。在共聚焦显微镜下,观察到轻链和重链在肾小球中的共定位。电子显微镜显示主要为上皮下颗粒状沉积物,无明显超微结构组织。所有患者均接受了类固醇治疗,观察到良好效果。1例患者进行的随访肾活检显示组织学改善。随访期间(平均44个月),无患者发生骨髓瘤或其他血液系统恶性肿瘤。
与膜性特征相关的MIDD是一种极其罕见但独特的实体。我们的研究提示非缺失的完整免疫球蛋白分子在肾小球沉积。该实体的患者似乎对类固醇治疗反应良好。