Literature DB >> 3500183

Z-type alpha 1-antitrypsin is less competent than M1-type alpha 1-antitrypsin as an inhibitor of neutrophil elastase.

F Ogushi1, G A Fells, R C Hubbard, S D Straus, R G Crystal.   

Abstract

Alpha 1-antitrypsin (alpha 1AT) deficiency resulting from homozygous inheritance of the Z-type alpha 1AT gene is associated with serum alpha 1AT levels of less than 50 mg/dl and the development of emphysema in the third to fourth decades. Despite the overwhelming evidence that the emphysema of PiZZ individuals develops because of a "deficiency" of alpha 1AT and hence an insufficient antineutrophil elastase defense of the lung, epidemiologic evidence has shown that levels of alpha 1AT of only 80 mg/dl protect the lung from an increased risk of emphysema. With this background, we hypothesized that homozygous inheritance of the Z-type may confer an added risk beyond a simple deficiency of alpha 1AT by virtue of an inability of the Z-type alpha 1AT molecule to inhibit neutrophil elastase as effectively as the common M1-type molecule. To evaluate this hypothesis, the functional status of alpha 1AT from PiZZ individuals (n = 10) was compared with that of alpha 1AT from PiM1M1 individuals (n = 7) for its ability to inhibit neutrophil elastase (percent inhibition) as well as its association rate constant for neutrophil elastase (K association). Plasma alpha 1AT concentration, measured by radial immunodiffusion, was 34 +/- 1 mg/dl in PiZZ patients vs. 237 +/- 14 mg/dl for PiM1M1 plasma, a sevenfold difference. When titrated against neutrophil elastase, the present inhibition of PiZZ plasma was significantly less than Pi M1M1 plasma (ZZ 78 +/- 1% vs. M1M1 95 +/- 1%, P less than 0.001) as was purified Z type alpha 1AT (ZZ, 63 +/- 2% vs. M1M1 86 +/- 2%, P less than 0.001). Sodium dodecyl sulfate (SDS) gel comparisons of the complexes formed with M1-type alpha 1AT and Z-type alpha 1AT with elastase demonstrated the Z alpha 1AT-elastase complexes were less stable than the M1 alpha 1AT-elastase complexes, thus releasing some of the enzyme to continue to function as a protease. Consistent with these observations, the K association of purified Z-type alpha 1AT for neutrophil elastase was lower than that of M1-type alpha 1AT (ZZ 4.5 +/- 0.3 X 10(6) M-1s-1 vs. M1M1 9.7 +/- 0.4 X 10(6) M-1s-1, P less than 0.001), suggesting that for the population of alpha 1AT molecules, the active Z-type molecules take more than twice as long as the active M1-type alpha 1AT to inhibit neutrophil elastase. Consequently, not only is there less alpha1AT in PiZZ individuals, but the population of Z-type alpha1AT molecules is less competent as an inhibitor of neutrophil elastase than M1-type alpha1AT molecules. This combination of defects suggests that PiZZ individuals have far less functional antielastase protection than suggested by the reduced concentrations of alpha1AT alone, further explaining their profound risk for development of emphysema.

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Year:  1987        PMID: 3500183      PMCID: PMC442392          DOI: 10.1172/JCI113214

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  70 in total

1.  Isolation and properties of human plasma alpha-1-proteinase inhibitor.

Authors:  R Pannell; D Johnson; J Travis
Journal:  Biochemistry       Date:  1974-12-17       Impact factor: 3.162

2.  Pathogenesis of deficient serum alpha1-antitrypsin in the type ZZ homozygote.

Authors:  P T Rowley; M L Sevilla; R H Schwartz
Journal:  Biochem Genet       Date:  1974-09       Impact factor: 1.890

3.  Serm alpha-antitrypsin types: elastase inhibition versus trypsin inhibition.

Authors:  P T Rowley; M L Sevilla; H Schwartz
Journal:  Hum Hered       Date:  1974       Impact factor: 0.444

4.  Induction of experimental emphysema in dogs using leukocyte homogenates.

Authors:  V Marco; B Mass; D R Meranze; G Weinbaum; P Kimbel
Journal:  Am Rev Respir Dis       Date:  1971-10

5.  Alpha-1-antitrypsin deficiency: a defect in protein synthesis.

Authors:  F Kueppers; R J Fallat
Journal:  Clin Chim Acta       Date:  1969-06       Impact factor: 3.786

6.  Purification and partial characterization of pas-positive inclusion bodies from the liver in alpha 1-antitrypsin deficiency.

Authors:  S Eriksson; C Larsson
Journal:  N Engl J Med       Date:  1975-01-23       Impact factor: 91.245

Review 7.  Alpha1-antitrypsin and its deficiency.

Authors:  F Kueppers; L F Black
Journal:  Am Rev Respir Dis       Date:  1974-08

8.  On the inhibition of elastase by serum. Some distinguishing properties of alpha1-antitrypsin and alpha2-macroglobulin.

Authors:  J F Meyer; J Bieth; P Metais
Journal:  Clin Chim Acta       Date:  1975-07-09       Impact factor: 3.786

9.  Purification of alpha1-antitrypsin from plasma through thiol-disulfide interchange.

Authors:  C B Laurell; J Pierce; U Persson; E Thulin
Journal:  Eur J Biochem       Date:  1975-09-01

10.  A community study of the relation of alpha1-antitrypsin levels to obstructive lung diseases.

Authors:  J O Morse; M D Lebowitz; R J Knudson; B Burrows
Journal:  N Engl J Med       Date:  1975-02-06       Impact factor: 91.245

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  55 in total

Review 1.  Alpha1-antitrypsin polymerization and the serpinopathies: pathobiology and prospects for therapy.

Authors:  David A Lomas; Ravi Mahadeva
Journal:  J Clin Invest       Date:  2002-12       Impact factor: 14.808

2.  How the serpin α1-proteinase inhibitor folds.

Authors:  Klavs Dolmer; Peter G W Gettins
Journal:  J Biol Chem       Date:  2012-02-13       Impact factor: 5.157

3.  Paucimannose-Rich N-glycosylation of Spatiotemporally Regulated Human Neutrophil Elastase Modulates Its Immune Functions.

Authors:  Ian Loke; Ole Østergaard; Niels H H Heegaard; Nicolle H Packer; Morten Thaysen-Andersen
Journal:  Mol Cell Proteomics       Date:  2017-06-19       Impact factor: 5.911

Review 4.  Genetics and respiratory disease. 2. Alpha 1-antitrypsin deficiency, cirrhosis and emphysema.

Authors:  R Mahadeva; D A Lomas
Journal:  Thorax       Date:  1998-06       Impact factor: 9.139

5.  Characterization of the gene and protein of the common alpha 1-antitrypsin normal M2 allele.

Authors:  T Nukiwa; M L Brantly; F Ogushi; G A Fells; R G Crystal
Journal:  Am J Hum Genet       Date:  1988-09       Impact factor: 11.025

Review 6.  Alpha 1-antitrypsin deficiency, emphysema, and liver disease. Genetic basis and strategies for therapy.

Authors:  R G Crystal
Journal:  J Clin Invest       Date:  1990-05       Impact factor: 14.808

7.  Fate of aerosolized recombinant DNA-produced alpha 1-antitrypsin: use of the epithelial surface of the lower respiratory tract to administer proteins of therapeutic importance.

Authors:  R C Hubbard; M A Casolaro; M Mitchell; S E Sellers; F Arabia; M A Matthay; R G Crystal
Journal:  Proc Natl Acad Sci U S A       Date:  1989-01       Impact factor: 11.205

8.  α₁-Antitrypsin protease inhibitor MZ heterozygosity is associated with airflow obstruction in two large cohorts.

Authors:  Inga-Cecilie Sørheim; Per Bakke; Amund Gulsvik; Sreekumar G Pillai; Ane Johannessen; Per I Gaarder; Edward J Campbell; Alvar Agustí; Peter M A Calverley; Claudio F Donner; Barry J Make; Stephen I Rennard; Jørgen Vestbo; Emiel F M Wouters; Peter D Paré; Robert D Levy; Harvey O Coxson; David A Lomas; Craig P Hersh; Edwin K Silverman
Journal:  Chest       Date:  2010-07-01       Impact factor: 9.410

9.  Conformational properties of the disease-causing Z variant of α1-antitrypsin revealed by theory and experiment.

Authors:  Itamar Kass; Anja S Knaupp; Stephen P Bottomley; Ashley M Buckle
Journal:  Biophys J       Date:  2012-06-19       Impact factor: 4.033

10.  A Pro----Leu substitution in codon 369 of the alpha-1-antitrypsin deficiency variant PI MHeerlen.

Authors:  M H Hofker; T Nukiwa; H M van Paassen; M Nelen; J A Kramps; E C Klasen; R R Frants; R G Crystal
Journal:  Hum Genet       Date:  1989-02       Impact factor: 4.132

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