Kato Tamayo, Ushiogi Yasuyuki, Yokoyama Hitoshi, Hara Shigeo, Matsunaga Akira, Muso Eri, Saito Takao
Department of Nephrology, Fukuiken Saiseikai Hospital, 7-1 Funabashi, Wadanakacho, Fukui, 918-8503, Japan.
Department of Nephrology, Kanazawa Medical University School of Medicine, Uchinada, Japan.
CEN Case Rep. 2019 May;8(2):106-111. doi: 10.1007/s13730-019-00380-w. Epub 2019 Jan 30.
A 47-year-old Japanese man with mild proteinuria was treated with an ACE inhibitor and antiplatelet agent for 7 years. However, urinary protein levels increased and renal biopsy was performed. Eight out of 20 glomeruli showed global or segmental sclerosis with foamy changes or bubbles, but with a different appearance to typical foam cells or lipoprotein thrombi. "Spike" formation, as observed in membranous nephropathy (MN), was segmentally detected in methenamine silver-stained sections. In an immunofluorescence study, weak linear patterns for IgG and scanty deposits for C3 were observed in glomeruli, but were not specific for immunogenetic MN. An electron microscopy study showed highly dense deposits in the subepithelial, subendothelial, and mesangial areas, in which microbubbles appeared under a higher magnification. Since this case exhibited hypertriglyceridemia and cholesterolemia with high serum apolipoprotein E (apoE) clinically and homozygous apoE2/2 by apoE phenotype and genotype analyses, apoE2 homozygote glomerulopathy was diagnosed and various lipid-lowering agents, e.g., probucol, fenofibrate, and ezetimibe, were administered. However, renal dysfunction gradually developed and peritoneal dialysis was initiated 11 years after the diagnosis. ApoE Toyonaka (Ser197Cys) and homozygous E2/2 were recently identified by direct DNA sequencing. Therefore, non-immune MN-like lesions may develop with the combination of these apoE mutations.
一名47岁的日本男性,有轻度蛋白尿,接受血管紧张素转换酶抑制剂和抗血小板药物治疗7年。然而,尿蛋白水平升高,遂进行了肾活检。20个肾小球中有8个显示全球性或节段性硬化,伴有泡沫样改变或气泡,但外观与典型泡沫细胞或脂蛋白血栓不同。在亚甲胺银染色切片中,可部分检测到在膜性肾病(MN)中观察到的“钉突”形成。免疫荧光研究显示,肾小球中IgG呈弱阳性线性模式,C3沉积稀少,但并非免疫遗传性MN所特有。电子显微镜研究显示,上皮下、内皮和系膜区有高度致密的沉积物,在更高放大倍数下可见微气泡。由于该病例临床显示高甘油三酯血症和胆固醇血症,血清载脂蛋白E(apoE)水平高,且经apoE表型和基因型分析为纯合子apoE2/2,故诊断为apoE2纯合子肾小球病,并给予了多种降脂药物,如普罗布考、非诺贝特和依折麦布。然而,肾功能逐渐恶化,诊断11年后开始进行腹膜透析。最近通过直接DNA测序鉴定出apoE丰中(Ser197Cys)和纯合子E2/2。因此,这些apoE突变的组合可能会导致非免疫性MN样病变。