Aumailley M, Pöschl E, Martin G R, Yamada Y, Müller P K
Laboratory of Developmental Biology and Anomalies, National Institute for Dental Research, Bethesda, MD 20205.
Eur J Clin Invest. 1988 Apr;18(2):207-12. doi: 10.1111/j.1365-2362.1988.tb02415.x.
The Ehlers-Danlos syndrome type IV represents a heterogeneous group of diseases, the molecular defect of which seems to reside in a defective synthesis and processing of collagen III. Here we present data concerning both protein and mRNA levels of collagens I and III in cell cultures established from skin of nine patients and six controls. All patients investigated were characterized by a reduced amount of procollagen III in fibroblast cultures. However, the levels of collagen III propeptides, measured by a radioimmunoassay in culture medium of fibroblasts from the patients, were either decreased or in the range of control levels. Using hybridization with cDNA probes specific for pro alpha 1 (I) and pro alpha 1 (III) collagen chains we determined the ratios of procollagens I and III mRNA. Although synthesis of procollagen III was markedly reduced in fibroblasts of all patients studied, no parallel decrease of procollagen III specific mRNA was found. However, two distinctive groups of patients were identified, one with an unaltered ratio of procollagen I/III mRNA and the other which had an even higher proportion of collagen III mRNA.
IV型埃勒斯-当洛综合征代表一组异质性疾病,其分子缺陷似乎在于III型胶原的合成和加工存在缺陷。在此,我们展示了从9名患者和6名对照者的皮肤建立的细胞培养物中I型和III型胶原的蛋白质及mRNA水平的数据。所有接受调查的患者在成纤维细胞培养物中均表现为前胶原III含量减少。然而,通过放射免疫测定法在患者成纤维细胞培养基中测得的III型胶原前肽水平要么降低,要么处于对照水平范围内。使用与I型前α1链和III型前α1链特异性的cDNA探针杂交,我们测定了前胶原I和III mRNA的比例。尽管在所有研究患者的成纤维细胞中前胶原III的合成明显减少,但未发现前胶原III特异性mRNA平行下降。然而,确定了两组不同的患者,一组前胶原I/III mRNA比例未改变,另一组胶原III mRNA比例更高。