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PMID: 1702098 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

A deletion in the gene for glycoprotein IIb associated with Glanzmann's thrombasthenia.

The Journal of clinical investigation ·Vol. 87 ·No. 1 ·1991-01-00 ·Pages 270-6

Burk CD, Newman PJ, Lyman S, Gill J, Coller BS, Poncz M

Abstract

The platelet fibrinogen receptor is composed of a complex of glycoproteins (GP) IIb and IIIa on the surface of platelets. Deficient function of this receptor prevents normal platelet aggregation, resulting in Glanzmann's thrombasthenia (GT). In this paper, we describe a black thrombasthenic patient who is either homozygous or hemizygous for a deletion within the GPIIb gene. Initial Western blot analysis of platelet proteins from this patient did not detect any GPIIb, but did detect small amounts of GPIIIa of normal mobility. Quantitation of vitronectin receptor (VNR) demonstrated that this thrombasthenic patient had approximately 1.5-2 times the number of these receptors per platelet compared with controls, a finding that has previously been noted in other thrombasthenic patients with defects in GPIIb. Genomic Southern blot studies demonstrated a deletion in the GPIIb gene of approximately 4.5 kilobasepairs (kb). Analysis of the isolated GPIIb gene demonstrated that the deletion begins between two Alu repeats within intron 1 and ends in intron 9. Polymerase chain reaction (PCR) studies using platelet RNA and oligonucleotides directed to both the 5' and 3' ends of the GPIIb cDNA sequence easily detected GPIIb transcript, suggesting that the genomic deletion of exons 2-9 does not significantly decrease the level of the GPIIb mRNA. Sequence analysis of PCR-generated GPIIb cDNA showed that a cryptic AG splice acceptor sequence was being utilized, resulting in a transcript that contained a portion of introns 1 and 9, as well as having a deletion of exons 2-9. Unlike the GPIIb gene, the GPIIIa gene appears to be intact by Southern blot analysis. PCR studies using platelet RNA and oligonucleotides directed to the GPIIIa cDNA sequence demonstrated the presence of GPIIIa mRNA. In summary, the thrombasthenic state in this patient appears to be due to a GPIIb gene deletion resulting in an abnormal transcript and no detectable platelet GPIIb. Platelet GPIIIa levels were secondarily low presumably due to the known instability of GPIIIa in the absence of GPIIb.

MeSH Terms
Adolescent Base Sequence Blood Platelets/chemistry Blotting, Southern Chromosome Deletion Humans Immunoblotting Male Molecular Sequence Data Platelet Membrane Glycoproteins/genetics Polymerase Chain Reaction Receptors, Immunologic/analysis Receptors, Vitronectin Thrombasthenia/genetics
Chemicals
Platelet Membrane Glycoproteins Receptors, Immunologic Receptors, Vitronectin
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Burk C D
Children's Hospital of Philadelphia, Department of Pediatrics, University of Pennsylvania School of Medicine 19104.
Newman P J
Lyman S
Gill J
Coller B S
Poncz M
References (54)
54 references, click to expand
  1. Chromosomal localization of the genes for the vitronectin and fibronectin receptors alpha subunits and for platelet glycoproteins IIb and IIIa.
    J Clin Invest. 1988 Jun;81(6):1993-8 PMID: 2454952
  2. Amino acid sequence of the alpha subunit of human leukocyte adhesion receptor Mo1 (complement receptor type 3).
    J Cell Biol. 1988 Jun;106(6):2153-8 PMID: 2454931
  3. Human endothelial cells synthesize and express an Arg-Gly-Asp-directed adhesion receptor involved in attachment to fibrinogen and von Willebrand factor.
    Proc Natl Acad Sci U S A. 1987 Sep;84(18):6471-5 PMID: 2442758
  4. Structure of the platelet membrane glycoprotein IIb. Homology to the alpha subunits of the vitronectin and fibronectin membrane receptors.
    J Biol Chem. 1987 Jun 25;262(18):8476-82 PMID: 2439501
  5. Biosynthetic and functional properties of an Arg-Gly-Asp-directed receptor involved in human melanoma cell attachment to vitronectin, fibrinogen, and von Willebrand factor.
    J Biol Chem. 1987 Dec 25;262(36):17703-11 PMID: 2447074
  6. Amino acid sequence of the vitronectin receptor alpha subunit and comparative expression of adhesion receptor mRNAs.
    J Biol Chem. 1987 Oct 15;262(29):14080-5 PMID: 2443500
  7. Immunochemical and amino-terminal sequence comparison of two cytoadhesins indicates they contain similar or identical beta subunits and distinct alpha subunits.
    J Biol Chem. 1987 Apr 25;262(12):5437-40 PMID: 2437107
  8. A novel integrin (alpha E beta 4) from human epithelial cells suggests a fourth family of integrin adhesion receptors.
    EMBO J. 1989 Mar;8(3):673-80 PMID: 2542022
  9. Efficient surface expression of platelet GPIIb-IIIa requires both subunits.
    Blood. 1989 Jul;74(1):14-8 PMID: 2752106
  10. Physical linkage of the genes for platelet membrane glycoproteins IIb and IIIa.
    Proc Natl Acad Sci U S A. 1988 Nov;85(22):8683-7 PMID: 3186752
  11. Thrombasthenia with an abnormal platelet membrane glycoprotein IIb of different molecular weight.
    Blood. 1988 Apr;71(4):915-22 PMID: 3355897
  12. Effects of the cell adhesion peptide, Arg-Gly-Asp-Ser, on responses of washed platelets from humans, rabbits, and rats.
    Blood. 1988 Jan;71(1):132-6 PMID: 3334894
  13. The silent carrier allele: beta thalassemia without a mutation in the beta-globin gene or its immediate flanking regions.
    Cell. 1984 Nov;39(1):123-8 PMID: 6091904
  14. cDNA cloning and complete primary structure of the alpha subunit of a leukocyte adhesion glycoprotein, p150,95.
    EMBO J. 1987 Dec 20;6(13):4023-8 PMID: 3327687
  15. LFA-1 immunodeficiency disease. Definition of the genetic defect and chromosomal mapping of alpha and beta subunits of the lymphocyte function-associated antigen 1 (LFA-1) by complementation in hybrid cells.
    J Exp Med. 1986 Sep 1;164(3):855-67 PMID: 3528378
  16. Protein sequence of endothelial glycoprotein IIIa derived from a cDNA clone. Identity with platelet glycoprotein IIIa and similarity to "integrin".
    J Biol Chem. 1987 Mar 25;262(9):3936-9 PMID: 3494014
  17. Type I Glanzmann thrombasthenia patients from the Iraqi-Jewish and Arab populations in Israel can be differentiated by platelet glycoprotein IIIa immunoblot analysis.
    Blood. 1987 Jun;69(6):1696-703 PMID: 3580574
  18. A variant of Glanzmann's thrombasthenia with abnormal glycoprotein IIb-IIIa complexes in the platelet membrane.
    J Clin Invest. 1987 Mar;79(3):962-9 PMID: 3818957
  19. Inhibition of platelet adhesion to fibronectin, fibrinogen, and von Willebrand factor substrates by a synthetic tetrapeptide derived from the cell-binding domain of fibronectin.
    Blood. 1985 Oct;66(4):946-52 PMID: 3876125
  20. Construction of human gene libraries from small amounts of peripheral blood: analysis of beta-like globin genes.
    Hemoglobin. 1982;6(1):27-36 PMID: 7068433
  21. "Nonrandom" DNA sequence analysis in bacteriophage M13 by the dideoxy chain-termination method.
    Proc Natl Acad Sci U S A. 1982 Jul;79(14):4298-302 PMID: 6956859
  22. PECAM-1 (CD31) cloning and relation to adhesion molecules of the immunoglobulin gene superfamily.
    Science. 1990 Mar 9;247(4947):1219-22 PMID: 1690453
  23. The Alu family of dispersed repetitive sequences.
    Science. 1982 Jun 4;216(4550):1065-70 PMID: 6281889
  24. Glanzmann thrombasthenia: deficient binding of von Willebrand factor to thrombin-stimulated platelets.
    Proc Natl Acad Sci U S A. 1982 Oct;79(19):6038-41 PMID: 6310559
  25. Inhibition of fibrinogen binding to stimulated human platelets by a monoclonal antibody.
    Proc Natl Acad Sci U S A. 1983 May;80(9):2417-21 PMID: 6302680
  26. Identification of the fibrinogen receptor on human platelets by photoaffinity labeling.
    J Biol Chem. 1982 Jul 25;257(14):8049-54 PMID: 6282870
  27. The primary structure of the VLA-2/collagen receptor alpha 2 subunit (platelet GPIa): homology to other integrins and the presence of a possible collagen-binding domain.
    J Cell Biol. 1989 Jul;109(1):397-407 PMID: 2545729
  28. The platelet membrane glycoprotein IIb-IIIa complex.
    Blood. 1988 Apr;71(4):831-43 PMID: 2833328
  29. New perspectives in cell adhesion: RGD and integrins.
    Science. 1987 Oct 23;238(4826):491-7 PMID: 2821619
  30. Organization of the gene for platelet glycoprotein IIb.
    Biochemistry. 1990 Feb 6;29(5):1232-44 PMID: 2322558
  31. Identification of an abnormal gene for the GPIIIa subunit of the platelet fibrinogen receptor resulting in Glanzmann's thrombasthenia.
    Blood. 1990 Feb 15;75(4):881-8 PMID: 1967954
  32. Glanzmann's thrombasthenia: the spectrum of clinical disease.
    Blood. 1990 Apr 1;75(7):1383-95 PMID: 2180491
  33. Polymorphism of human platelet membrane glycoprotein IIb associated with the Baka/Bakb alloantigen system.
    Blood. 1990 Jun 15;75(12):2343-8 PMID: 2350579
  34. Cloning of the beta subunit of the leukocyte adhesion proteins: homology to an extracellular matrix receptor defines a novel supergene family.
    Cell. 1987 Feb 27;48(4):681-90 PMID: 3028646
  35. Integrins: a family of cell surface receptors.
    Cell. 1987 Feb 27;48(4):549-54 PMID: 3028640
  36. The Drosophila PS2 antigen is an invertebrate integrin that, like the fibronectin receptor, becomes localized to muscle attachments.
    Cell. 1987 Dec 24;51(6):929-40 PMID: 2961459
  37. The tetrapeptide analogue of the cell attachment site of fibronectin inhibits platelet aggregation and fibrinogen binding to activated platelets.
    J Biol Chem. 1985 Oct 5;260(22):11891-4 PMID: 2995350
  38. Synthesis by cultured human umbilical vein endothelial cells of two proteins structurally and immunologically related to platelet membrane glycoproteins IIb and IIIa.
    J Cell Biol. 1986 Jul;103(1):81-6 PMID: 2941443
  39. Residual amounts of glycoproteins IIb and IIIa may be present in the platelets of most patients with Glanzmann's thrombasthenia.
    Blood. 1985 Apr;65(4):1021-4 PMID: 3156640
  40. Quantitation of membrane glycoprotein IIIa on intact human platelets using the monoclonal antibody, AP-3.
    Blood. 1985 Jan;65(1):227-32 PMID: 3155488
  41. Amino acid sequence of the human fibronectin receptor.
    J Cell Biol. 1987 Sep;105(3):1183-90 PMID: 2958481
  42. Amino acid sequence of the murine Mac-1 alpha chain reveals homology with the integrin family and an additional domain related to von Willebrand factor.
    EMBO J. 1988 May;7(5):1371-8 PMID: 3044779
  43. Biosynthesis and assembly of platelet GPIIb-IIIa in human megakaryocytes: evidence that assembly between pro-GPIIb and GPIIIa is a prerequisite for expression of the complex on the cell surface.
    Blood. 1989 Oct;74(5):1603-11 PMID: 2477081
  44. A novel vitronectin receptor integrin (alpha v beta x) is responsible for distinct adhesive properties of carcinoma cells.
    Cell. 1989 Apr 7;57(1):59-69 PMID: 2467745
  45. Isolation and characterization of a platelet membrane protein related to the vitronectin receptor.
    J Biol Chem. 1989 Mar 5;264(7):3742-9 PMID: 2465293
  46. Comparison of cDNA-derived protein sequences of the human fibronectin and vitronectin receptor alpha-subunits and platelet glycoprotein IIb.
    Biochemistry. 1987 Dec 15;26(25):8158-65 PMID: 2450560
  47. Purification of glycoproteins IIb and III from human platelet plasma membranes and characterization of a calcium-dependent glycoprotein IIb-III complex.
    J Biol Chem. 1982 Sep 10;257(17):10458-66 PMID: 6213621
  48. The formation of Ca++-dependent complexes of platelet membrane glycoproteins IIb and IIIa in solution as determined by crossed immunoelectrophoresis.
    Blood. 1981 Aug;58(2):268-78 PMID: 6454451
  49. Reduced surface expression and binding of fibronectin by thrombin-stimulated thrombasthenic platelets.
    J Clin Invest. 1983 Mar;71(3):619-24 PMID: 6826726
  50. DNA sequencing with chain-terminating inhibitors.
    Proc Natl Acad Sci U S A. 1977 Dec;74(12):5463-7 PMID: 271968
  51. Cloning of glycoprotein IIIa cDNA from human erythroleukemia cells and localization of the gene to chromosome 17.
    Blood. 1988 Aug;72(2):593-600 PMID: 3165296
  52. Structure of platelet glycoprotein IIIa. A common subunit for two different membrane receptors.
    J Clin Invest. 1988 May;81(5):1470-5 PMID: 2452834
  53. An integrin receptor on normal and thrombasthenic platelets that binds thrombospondin.
    Blood. 1989 Nov 1;74(6):2022-7 PMID: 2478219
  54. Molecular cloning and chemical synthesis of a region of platelet glycoprotein IIb involved in adhesive function.
    Proc Natl Acad Sci U S A. 1987 Oct;84(20):7114-8 PMID: 2444974
Article Info
Journal
The Journal of clinical investigation
Abbr.
J Clin Invest
ISSN
0021-9738
Published
1991-01-00
Pages
270-6
Language
English
Region
United States
NLM ID
7802877
PMCID
PMC295042
Subset
IM
Grants
NHLBI NIH HHS · HL-07439 · United States
NHLBI NIH HHS · HL-38166 · United States
NHLBI NIH HHS · HL-40387 · United States
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M63429, M65091, M65092, M65093, M65094, M65095, M65096, M65097, M65098, M65099
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