Sbar G D, Venkataseshan V S, Huang Z, Marquet E, Brunswick J W, Churg J
Department of Pathology, Barnert Hospital, Paterson, N.J. 07514, USA.
Am J Nephrol. 1996;16(4):320-6. doi: 10.1159/000169017.
We encountered 4 individuals with Marfan syndrome who presented with microhematuria and proteinuria. In 2 of them, a renal biopsy was performed. The predominant glomerular change by light microscopy was a focal segmental increase in mesangial matrix with early sclerotic lesions. Ultrastructurally, there was variable subendothelial widening containing haphazardly arranged microfibrils, 10-13 nm in diameter. Changes in small arteries present in the biopsies were mild in case 1 and more striking in case 2 which consisted of elastolysis and fragmentation and focal disruption of internal elastic lamina, and focal degenerative changes in the media. In light of observations on the abnormalities of microfibrillar protein (fibrillin) in the microfibrillar-fiber system and the presence of abnormal type IV collagen in the connective tissues in Marfan syndrome, the glomerular basement membrane alterations may be related to these defects and lead to microhematuria and proteinuria.
我们遇到了4例患有马凡综合征且出现镜下血尿和蛋白尿的患者。其中2例进行了肾活检。光镜下肾小球的主要变化是系膜基质局灶节段性增加并伴有早期硬化性病变。超微结构上,可见内皮下可变增宽,其中含有直径为10 - 13 nm的杂乱排列的微原纤维。活检中存在的小动脉变化在病例1中较轻,在病例2中更明显,包括弹性纤维溶解和断裂以及内弹性膜的局灶性破坏,以及中膜的局灶性退行性改变。鉴于对马凡综合征微原纤维-纤维系统中微原纤维蛋白(原纤蛋白)异常以及结缔组织中异常IV型胶原存在的观察,肾小球基底膜改变可能与这些缺陷有关,并导致镜下血尿和蛋白尿。