Spotila L D, Constantinou C D, Sereda L, Ganguly A, Riggs B L, Prockop D J
Department of Biochemistry and Molecular Biology, Jefferson Institute of Molecular Medicine, Jefferson Medical College, Philadelphia, PA 19107-6799.
Proc Natl Acad Sci U S A. 1991 Jun 15;88(12):5423-7. doi: 10.1073/pnas.88.12.5423.
Mutations in the two genes for type I collagen (COL1A1 or COL1A2) cause osteogenesis imperfecta (OI), a heritable disease characterized by moderate to extreme brittleness of bone early in life. Here we show that a 52-year-old postmenopausal woman with severe osteopenia and a compression fracture of a thoracic vertebra had a mutation in the gene for the alpha 2(I) chain of type I collagen (COL1A2) similar to mutations that cause OI. cDNA was prepared from the woman's skin fibroblast RNA and assayed for the presence of a mutation by treating DNA heteroduplexes with carbodiimide. The results indicated a sequence variation in the region encoding amino acid residues 660-667 of the alpha 2(I) chain. Further analysis demonstrated a single-base mutation that caused a serine-for-glycine substitution at position 661 of the alpha 2(I) triple-helical domain. The substitution produced posttranslational overmodification of the collagen triple helix, as is seen with most glycine substitutions that cause OI. The patient had a history of five previous fractures, slightly blue sclerae, and slight hearing loss. Therefore, the results suggest that there may be phenotypic and genotypic overlap between mild osteogenesis imperfecta and postmenopausal osteoporosis, and that a subset of women with postmenopausal osteoporosis may have mutations in the genes for type I procollagen.
I型胶原蛋白(COL1A1或COL1A2)的两个基因发生突变会导致成骨不全症(OI),这是一种遗传性疾病,其特征是在生命早期骨骼具有中度至极度脆性。我们在此报告,一名52岁的绝经后女性患有严重骨质减少和胸椎压缩性骨折,其I型胶原蛋白α2(I)链基因(COL1A2)发生了与导致OI的突变类似的突变。从该女性的皮肤成纤维细胞RNA制备cDNA,并通过用碳二亚胺处理DNA异源双链体来检测是否存在突变。结果表明在编码α2(I)链氨基酸残基660 - 667的区域存在序列变异。进一步分析显示一个单碱基突变导致α2(I)三螺旋结构域第661位的丝氨酸取代甘氨酸。这种取代导致胶原蛋白三螺旋的翻译后过度修饰,这与大多数导致OI的甘氨酸取代情况相同。该患者有过五次骨折史、轻度蓝巩膜和轻度听力损失。因此,结果表明轻度成骨不全症和绝经后骨质疏松症之间可能存在表型和基因型重叠,并且一部分绝经后骨质疏松症女性可能在I型前胶原基因中存在突变。