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一名患有绝经后骨质疏松症女性的I型前胶原基因(COL1A2)突变:与轻度成骨不全症表型和基因型重叠的证据

Mutation in a gene for type I procollagen (COL1A2) in a woman with postmenopausal osteoporosis: evidence for phenotypic and genotypic overlap with mild osteogenesis imperfecta.

作者信息

Spotila L D, Constantinou C D, Sereda L, Ganguly A, Riggs B L, Prockop D J

机构信息

Department of Biochemistry and Molecular Biology, Jefferson Institute of Molecular Medicine, Jefferson Medical College, Philadelphia, PA 19107-6799.

出版信息

Proc Natl Acad Sci U S A. 1991 Jun 15;88(12):5423-7. doi: 10.1073/pnas.88.12.5423.

DOI:10.1073/pnas.88.12.5423
PMID:2052622
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC51885/
Abstract

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.

摘要

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。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/ac67f418ac0e/pnas01062-0369-d.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/5a5f8793168f/pnas01062-0368-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/86e5537e297c/pnas01062-0368-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/27fb9971ebd0/pnas01062-0368-c.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/cf8229133d21/pnas01062-0369-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/ef019f773eae/pnas01062-0369-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/34a058b293e0/pnas01062-0369-c.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/ac67f418ac0e/pnas01062-0369-d.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/5a5f8793168f/pnas01062-0368-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/86e5537e297c/pnas01062-0368-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/27fb9971ebd0/pnas01062-0368-c.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/cf8229133d21/pnas01062-0369-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/ef019f773eae/pnas01062-0369-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/34a058b293e0/pnas01062-0369-c.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fd4/51885/ac67f418ac0e/pnas01062-0369-d.jpg

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本文引用的文献

1
A simple and very efficient method for generating cDNA libraries.一种简单且非常有效的生成cDNA文库的方法。
Gene. 1983 Nov;25(2-3):263-9. doi: 10.1016/0378-1119(83)90230-5.
2
Nuclease S1 mapping of a homozygous mutation in the carboxyl-propeptide-coding region of the pro alpha 2(I) collagen gene in a patient with osteogenesis imperfecta.对一名成骨不全患者的原α2(I)型胶原蛋白基因羧基前肽编码区纯合突变进行核酸酶S1图谱分析。
Proc Natl Acad Sci U S A. 1984 Jul;81(14):4524-8. doi: 10.1073/pnas.81.14.4524.
3
Involutional osteoporosis.老年性骨质疏松症
番茄红素在预防绝经后骨丢失中的作用:从分子到临床研究的证据。
Int J Mol Sci. 2020 Sep 27;21(19):7119. doi: 10.3390/ijms21197119.
4
Special form of osteoporosis in a 53-year-old man.一名53岁男性骨质疏松症的特殊形式。
BMJ Case Rep. 2018 Dec 13;11(1):e226672. doi: 10.1136/bcr-2018-226672.
5
Genetics of osteoporosis: searching for candidate genes for bone fragility.骨质疏松症的遗传学:寻找骨脆性的候选基因。
Arch Endocrinol Metab. 2016 Aug;60(4):391-401. doi: 10.1590/2359-3997000000178.
6
Atypical femoral fracture following bisphosphonate treatment in a woman with osteogenesis imperfecta--a case report.一名患有成骨不全症的女性在接受双膦酸盐治疗后发生非典型股骨骨折——病例报告
Acta Orthop. 2012 Oct;83(5):548-50. doi: 10.3109/17453674.2012.729183. Epub 2012 Sep 24.
7
Perspective on post-menopausal osteoporosis: establishing an interdisciplinary understanding of the sequence of events from the molecular level to whole bone fractures.绝经后骨质疏松症的研究进展:从分子水平到整个骨骨折事件序列的建立跨学科理解。
J R Soc Interface. 2010 Mar 6;7(44):353-72. doi: 10.1098/rsif.2009.0282. Epub 2009 Oct 21.
8
Variable bone fragility associated with an Amish COL1A2 variant and a knock-in mouse model.与阿什肯纳兹 COL1A2 变异体和敲入小鼠模型相关的可变骨脆性。
J Bone Miner Res. 2010 Feb;25(2):247-61. doi: 10.1359/jbmr.090720.
9
Bone fragility and collagen cross-links.骨脆性与胶原蛋白交联
J Bone Miner Res. 2004 Dec;19(12):2000-4. doi: 10.1359/JBMR.040820. Epub 2004 Aug 30.
10
Gene expression studies of osteoporosis: implications for microarray research.骨质疏松症的基因表达研究:对微阵列研究的启示
Osteoporos Int. 2003 Jul;14(6):451-61. doi: 10.1007/s00198-002-1373-0. Epub 2003 Apr 29.
N Engl J Med. 1986 Jun 26;314(26):1676-86. doi: 10.1056/NEJM198606263142605.
4
Characterization of DNA sequences associated with residual nuclei of Saccharomyces cerevisiae.与酿酒酵母残余细胞核相关的DNA序列的表征
Exp Cell Res. 1986 Jul;165(1):29-40. doi: 10.1016/0014-4827(86)90530-6.
5
Structure of a full-length cDNA clone for the prepro alpha 2(I) chain of human type I procollagen. Comparison with the chicken gene confirms unusual patterns of gene conservation.人I型前胶原α2(I)链前体全长cDNA克隆的结构。与鸡基因的比较证实了基因保守性的异常模式。
Biochem J. 1988 Jun 15;252(3):633-40. doi: 10.1042/bj2520633.
6
Osteogenesis imperfecta. A model for genetic causes of osteoporosis and perhaps several other common diseases of connective tissue.成骨不全症。一种骨质疏松症以及可能其他几种常见结缔组织疾病遗传病因的模型。
Arthritis Rheum. 1988 Jan;31(1):1-8. doi: 10.1002/art.1780310101.
7
A substitution of cysteine for glycine 748 of the alpha 1 chain produces a kink at this site in the procollagen I molecule and an altered N-proteinase cleavage site over 225 nm away.
J Biol Chem. 1988 Dec 15;263(35):19249-55.
8
Enzymatic amplification of beta-globin genomic sequences and restriction site analysis for diagnosis of sickle cell anemia.用于镰状细胞贫血诊断的β-珠蛋白基因组序列的酶促扩增及限制性酶切位点分析。
Science. 1985 Dec 20;230(4732):1350-4. doi: 10.1126/science.2999980.
9
Reduced bone mass in daughters of women with osteoporosis.骨质疏松症女性的女儿骨量减少。
N Engl J Med. 1989 Mar 2;320(9):554-8. doi: 10.1056/NEJM198903023200903.
10
Osteoporosis and familial idiopathic scoliosis: association with an abnormal alpha 2(I) collagen.
Connect Tissue Res. 1989;21(1-4):117-23; discussion 124. doi: 10.3109/03008208909050002.