Amino acid analyses were carried out on dentin proteins obtained from 33 normal teeth and 59 teeth from osteogenesis imperfecta patients. The analyses revealed that in the control teeth approximately 95% of the insoluble dentin fraction was collagen. The majority of the analyses for teeth obtained from the O.I. patients were biochemically abnormal (55 out of 59). Specifically, there was a significant increase in the acidic amino acids, with a corresponding decrease in the basic amino acids. A small group of patients showed double peaks in the histidine, hydroxylysine, and lysine areas of the chromatograms. These results emphasize that despite the fact that the teeth of O.I. patients may appear to be clinically normal, all but four of the teeth examined had abnormal dentin collagen.
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References
1.
Barsh, G.S.; David, K.E.; and Byers, P.H. (1982): Type I Osteogenesis Imperfecta-a Nonfunctional Allele for Pro α-1(I) Chains of Type I Procollagen, Proc Natl Acad Sci U.S.A. 79 :3838-3842.
2.
Bornstein, P. and Sage, H. (1980): Structurally Distinct Collagen Types, Ann Rev Biochem49:957-1003.
3.
Bornstein, P. and Traub, W. (1979): The Chemistry and Biology of Collagen. In: The Proteins, H. Neurath and R.L. Hiln, Eds., New York: Academic Press, pp. 411-632.
4.
Byers, P.H.; Bonadio, J.F.; and Steinmann, B. (1984): Osteogenesis Imperfecta: Update and Perspective , Am J Med Genet17:429-435.
5.
Byers, P.H.; Shapiro, J.R.; Rowe, D.W.; David, K.E.; and Holbrook, K.A. (1983): Abnormal α2 Chain Type I Collagen from a Patient with a Form of Osteogenesis Imperfecta, J Clin Invest71:689-710.
6.
Epstein, E.H., Jr.; Scott, R.D.; Miller, E.J.; and Piez, K.A. (1971): Isolation and Characterization of the Peptides Derived from Soluble Human and Baboon Skin Collagen after Cyanogen Bromide Cleavage, J Biol Chem246:1718-1724.
7.
Eastoe, J.E. and Leach, A.A. (1958): In: Recent Advances in Gelatin and Glue Research , G. Stainsby, Ed., London: Pergamon Press, pp. 173-178.
8.
Eastoe, J.E.; Martin, P.; and Thomas, N.R. (1973): The Amino Acid Composition of Human Hard Tissue Collagens in Osteogenesis Imperfecta and Dentinogenesis Imperfecta, Calcif Tissue Res12:91-100.
9.
Fietzek, P. and Kuhn, K. (1975): Information Contained in the Amino Acid Sequence of the 1(1) Chain and its Consequence upon the Formation of the Triple Helix of Fibrils and Cross-links, Mol Cell Biochem8:141-165.
10.
Francis, M.J.O. and Duksin, D. (1983): Heritable Disorders of Collagen Metabolism, Trends Biochem Sci8:231-234.
11.
Francis, M.J.O.; Williams, K.J.; Sykes, B.C.; and Smith, R. (1981): The Ratio of Collagen α1(I), α2 and α1 (III) Chains in the Skin of 31 Patients with Osteogenesis Imperfecta, Clin Sci60:617-623.
12.
Fujimoto, D.; Hirama, M.; and Iwashita, T. (1982): Histidinoalanine : A New Cross-linking Amino Acid in Calcified Tissue Collagen, Biochem Biophys Res Commun104:1107-1113.
13.
Gage, J.P. (1984): Electrophoretic Characterization of Peptides from Normal Human Dentine, Arch Oral Biol29:575-580.
14.
Hanauske-Abel, H.H. and Rohm, K.H. (1980): The Collagenous Part of Clq is Unaffected in the Hydroxylysine Deficient Disease, FEBS Letts110:73-76.
15.
Kuboki, Y.; Fujisawa, R.; Tsuzaki, M.; Fanglui, C.; and Sasaki, S. (1984): Presence of Lysinoalanine and Histidinoalanine in Bovine Dentine Phosphoprotein, Calcif Tissue Int36:126-128.
16.
Leaver, A.G. (1981): Non-collagenous Proteins in Bone. In: The Chemistry and Biology of Mineralised Connective Tissues, A. Veis, Ed., Amsterdam: Elsevier, pp. 323-326.
17.
Moore, S. (1963): On the Determination of Cysteine as Cysteic Acid , J Biol Chem238:235-237.
18.
Munksgaard, E.C. and Larsen, E. (1981): Isolation of Serine Rich Low Molecular Weight Protein from Odontoblast Cultures. In: The Chemistry and Biology of Mineralised Connective Tissues, A. Veis, Ed., Amsterdam: Elsevier, pp. 433-437.
19.
Nimni, M.E. (1983): Collagen Structure, Function and Metabolism in Normal and Fibrotic Tissues, Seminars in Arthritis and Rheumatism13:1-83.
20.
Paterson, C.R.; McAllison, S.; and Miller, R. (1983): Heterogeneity of Osteogenesis Imperfecta, Type I, J Med Genet20 :203-205.
21.
Petrovic, O.M. and Miller, E.J. (1984): An Unusual Pattern of Peptide-bound Lysine Metabolism in Collagen from an Infant with Lethal Perinatal Osteogenesis Imperfecta, J Clin Invest73:1569-1575.
22.
Piez, K.A. (1967): Soluble Collagen and the Components Resulting from its Denaturation. In: Treatise on Collagen, Vol. 1, G.N. Ramachandran, Ed., New York: Academic Press, pp. 208-252.
23.
Pinnell, S.R.; Krane, S.M.; Kenzora, J.E.; and Glimcher, M.J. (1972): A Heritable Disorder of Connective Tissue: Hydroxylysine Deficient Collagen Disease, New Engl J Med286:1013-1020.
24.
Prockop, D.J. (1984): Osteogenesis Imperfecta: Phenotypic Heterogeneity, Protein Suicide, Short and Long Collagen, Am J Human Genet36:499-505.
25.
Sillence, D.O. (1981): Osteogenesis Imperfecta: an Expanding Panorama of Varients, Clin Orthop Rel Res159:11-25.
26.
Smith, R.; Francis, M.J.O.; and Houghton, G.R. (1983): The Brittle Bone Syndrome: Osteogenesis Imperfecta , London: Butterworths & Co. Ltd., pp. 1-218.
27.
Termine, J.D.; Robery, P.G.; Fisher, L.W.; Shimokawa, H.; Drum, M.A.; Cohn, K.M.; Hawkins, G.R.; Cruz, J.B.; and Thompson, K.G. (1984): Osteonectin, Bone Proteoglycan and Phosphophoryn Defects in a Form of Bovine Osteogenesis Imperfecta, Proc Natl Acad Sci U.S .A. 81:2213-2217.
28.
Tietelbaum, S.L.; Kraft, W.J.; Lang, R.; and Avioli, L.V. (1974): Bone Collagen Aggregation Abnormalities in Osteogenesis Imperfecta, Calcif Tissue Res17:75-79.
29.
Veis, A. and Schleuter, R.J. (1964): The Macromolecular Organisation of Dentine Matrix CollagenI. Characterisation of Dentine Collagen, Biochem3:1650-1657.
30.
Walters, W. and Eyre, D.R. (1983): Collagen Cross-links in Human Dentin: Increasing Content of Hydroxypyridinium Residues with Age, Calcif Tissue Int35:401-405.
