Literature DB >> 2052622

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.

L D Spotila1, C D Constantinou, L Sereda, A Ganguly, B L Riggs, D J Prockop.   

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.

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Year:  1991        PMID: 2052622      PMCID: PMC51885          DOI: 10.1073/pnas.88.12.5423

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  17 in total

1.  Osteogenesis imperfecta. A model for genetic causes of osteoporosis and perhaps several other common diseases of connective tissue.

Authors:  D J Prockop
Journal:  Arthritis Rheum       Date:  1988-01

2.  Enzymatic amplification of beta-globin genomic sequences and restriction site analysis for diagnosis of sickle cell anemia.

Authors:  R K Saiki; S Scharf; F Faloona; K B Mullis; G T Horn; H A Erlich; N Arnheim
Journal:  Science       Date:  1985-12-20       Impact factor: 47.728

3.  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.

Authors:  H Kuivaniemi; G Tromp; M L Chu; D J Prockop
Journal:  Biochem J       Date:  1988-06-15       Impact factor: 3.857

Review 4.  Involutional osteoporosis.

Authors:  B L Riggs; L J Melton
Journal:  N Engl J Med       Date:  1986-06-26       Impact factor: 91.245

5.  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.

Authors:  B E Vogel; R Doelz; K E Kadler; Y Hojima; J Engel; D J Prockop
Journal:  J Biol Chem       Date:  1988-12-15       Impact factor: 5.157

6.  Reduced bone mass in daughters of women with osteoporosis.

Authors:  E Seeman; J L Hopper; L A Bach; M E Cooper; E Parkinson; J McKay; G Jerums
Journal:  N Engl J Med       Date:  1989-03-02       Impact factor: 91.245

7.  A simple and very efficient method for generating cDNA libraries.

Authors:  U Gubler; B J Hoffman
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Review 8.  Mutations in collagen genes: causes of rare and some common diseases in humans.

Authors:  H Kuivaniemi; G Tromp; D J Prockop
Journal:  FASEB J       Date:  1991-04       Impact factor: 5.191

9.  Substitution of serine for alpha 1(I)-glycine 844 in a severe variant of osteogenesis imperfecta minimally destabilizes the triple helix of type I procollagen. The effects of glycine substitutions on thermal stability are either position of amino acid specific.

Authors:  M Pack; C D Constantinou; K Kalia; K B Nielsen; D J Prockop
Journal:  J Biol Chem       Date:  1989-11-25       Impact factor: 5.157

10.  Characterization of DNA sequences associated with residual nuclei of Saccharomyces cerevisiae.

Authors:  J A Potashkin; J A Huberman
Journal:  Exp Cell Res       Date:  1986-07       Impact factor: 3.905

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  25 in total

1.  Genetic and environmental factors affecting bone mineral density in large families.

Authors:  S S Yeap; M Beaumont; A Bennett; N A Keating; D A White; D J Hosking
Journal:  Postgrad Med J       Date:  1998-06       Impact factor: 2.401

2.  Special form of osteoporosis in a 53-year-old man.

Authors:  Simon Lampart; Silvia Azzarello-Burri; Christoph Henzen; Stefan Fischli
Journal:  BMJ Case Rep       Date:  2018-12-13

Review 3.  Searching for gene defects that cause high bone mass.

Authors:  M P Whyte
Journal:  Am J Hum Genet       Date:  1997-06       Impact factor: 11.025

4.  Genetics and osteoporosis: role of the vitamin D receptor gene.

Authors:  P J Kelly; N Morrison; P N Sambrook; J A Eisman
Journal:  Agents Actions       Date:  1994-08

5.  Conformation-sensitive gel electrophoresis for rapid detection of single-base differences in double-stranded PCR products and DNA fragments: evidence for solvent-induced bends in DNA heteroduplexes.

Authors:  A Ganguly; M J Rock; D J Prockop
Journal:  Proc Natl Acad Sci U S A       Date:  1993-11-01       Impact factor: 11.205

6.  Genetics of osteoporosis.

Authors:  M L Brandi; M L Bianchi; J A Eisman; F Glorieux; S Adami; C E Fiore; R Nuti; S Ortolani
Journal:  Calcif Tissue Int       Date:  1994-09       Impact factor: 4.333

Review 7.  Gene expression studies of osteoporosis: implications for microarray research.

Authors:  V Dvornyk; R R Recker; H-W Deng
Journal:  Osteoporos Int       Date:  2003-04-29       Impact factor: 4.507

8.  Defective splicing of mRNA from one COL1A1 allele of type I collagen in nondeforming (type I) osteogenesis imperfecta.

Authors:  M L Stover; D Primorac; S C Liu; M B McKinstry; D W Rowe
Journal:  J Clin Invest       Date:  1993-10       Impact factor: 14.808

9.  Phenotypic variability and incomplete penetrance of spontaneous fractures in an inbred strain of transgenic mice expressing a mutated collagen gene (COL1A1).

Authors:  R Pereira; K Halford; B P Sokolov; J S Khillan; D J Prockop
Journal:  J Clin Invest       Date:  1994-04       Impact factor: 14.808

10.  Transgenic mice expressing a partially deleted gene for type I procollagen (COL1A1). A breeding line with a phenotype of spontaneous fractures and decreased bone collagen and mineral.

Authors:  R Pereira; J S Khillan; H J Helminen; E L Hume; D J Prockop
Journal:  J Clin Invest       Date:  1993-02       Impact factor: 14.808

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