Pan X, Chen N, Chen X
Department of Nephrology, Ruijin Hospital, Shanghai Second Medical University.
Zhonghua Nei Ke Za Zhi. 1997 Dec;36(12):812-5.
To observe the distribution of type IV collagen chains within basement membrane in patients with Alport syndrome (AS), discuss the pathogenesis of AS and assess the diagnostic value of indirect immunofluorescent study of type IV collagen chains. By using indirect immunofluorescence technique, the distribution of different chains of type IV collagen in specimen of renal (6 specimens) and skin (5 specimens) basement membrane of 8 AS patients belonging to 7 kindreds were investigated. In 4 male patients with X-linked dominant (XD) AS, the specific monoclonal antibodies against NC1 domains of the alpha 3, alpha 4 and alpha 5 chains of type IV collagen failed to localize on glomerular basement membrane (GBM) and the alpha 5 chain of type IV collagen were also absent in the epidermal basement membrane (EBM). The results of immunofluorescent study were positive in 3 autosomal dominant and 1 autosomal recessive AS patients as same as that in control. AS presents a biochemical abnormality of type IV collagen. The pathogenesis of XD-AS may associate with the abnormal products of alpha 5 chain. The immunofluorescent study of type IV collagen chains distribution within GBM and EBM by using monoclonal antibodies is useful in confirming the diagnosis of AS, especially XD-AS.
观察Alport综合征(AS)患者基底膜中IV型胶原链的分布,探讨AS的发病机制,并评估IV型胶原链间接免疫荧光研究的诊断价值。采用间接免疫荧光技术,对7个家系的8例AS患者肾组织(6例标本)和皮肤组织(5例标本)基底膜中IV型胶原不同链的分布进行研究。在4例X连锁显性(XD)AS男性患者中,针对IV型胶原α3、α4和α5链NC1结构域的特异性单克隆抗体未能定位在肾小球基底膜(GBM)上,且表皮基底膜(EBM)中也缺乏IV型胶原α5链。3例常染色体显性和1例常染色体隐性AS患者的免疫荧光研究结果与对照组相同,均为阳性。AS存在IV型胶原的生化异常。XD-AS的发病机制可能与α5链的异常产物有关。利用单克隆抗体对GBM和EBM中IV型胶原链分布进行免疫荧光研究,有助于确诊AS,尤其是XD-AS。