Polymers of alpha(1)-antitrypsin are chemotactic for human neutrophils: a new paradigm for the pathogenesis of emphysema.

Plasma deficiency of alpha(1)-antitrypsin is most commonly due to the Z mutation ((342)Glu--> Lys) and is associated with early-onset panlobular emphysema. The lung disease in these patients is attributed to the relative deficiency of circulating alpha(1)-antitrypsin resulting in uncontrolled neutrophil-derived proteolytic activity. We have previously demonstrated that the local deficiency of Z alpha(1)-antitrypsin is exacerbated by the formation of polymers within the lung and now show that this polymerization not only inactivates alpha(1)-antitrypsin but also converts the molecule to a chemoattractant for human neutrophils. The chemotactic action of polymeric alpha(1)-antitrypsin was substantially greater than that seen with other conformers, was of similar magnitude to C5a, and was apparent over a range of physiologically relevant concentrations (EC(50) 0.0045 +/- 0.002 mg/ml). The biologic activity of polymeric alpha(1)-antitrypsin was confirmed by the demonstration that polymers, but not native alpha(1)-antitrypsin, induced neutrophil shape change and stimulated myeloperoxidase release and neutrophil adhesion. Polymeric alpha(1)-antitrypsin had no effect on basal or N-formyl-Met-Leu-Phe- stimulated superoxide anion release or constitutive apoptosis. The chemotactic properties of polymeric alpha(1)-antitrypsin may provide an explanation for the excessive neutrophils found in the lungs of Z alpha(1)-antitrypsin homozygotes and suggests a new paradigm for the pathogenesis of emphysema in these patients.

[1]  A. Klokke,et al.  Relationship between Weber-Christian panniculitis and the ZZ phenotype of alpha1-antitrypsin , 2004, Archives of Dermatological Research.

[2]  D. Lomas,et al.  Pathogenic α1-Antitrypsin Polymers Are Formed by Reactive Loop-β-Sheet A Linkage* , 2000, The Journal of Biological Chemistry.

[3]  R. Stockley,et al.  Airways inflammation in chronic bronchitis: the effects of smoking and alpha1-antitrypsin deficiency. , 2000, The European respiratory journal.

[4]  D. Lomas,et al.  A Kinetic Mechanism for the Polymerization of α1-Antitrypsin* , 1999, The Journal of Biological Chemistry.

[5]  D. Lomas,et al.  Heteropolymerization of S, I, and Z α1-antitrypsin and liver cirrhosis , 1999 .

[6]  F. Dallegri,et al.  Soluble Fas ligand is chemotactic for human neutrophilic polymorphonuclear leukocytes. , 1999, Journal of immunology.

[7]  E. Piitulainen,et al.  Decline in FEV1 related to smoking status in individuals with severe alpha1-antitrypsin deficiency (PiZZ). , 1999, The European respiratory journal.

[8]  D. Lomas,et al.  Lung polymers in Z alpha1-antitrypsin deficiency-related emphysema. , 1998, American journal of respiratory cell and molecular biology.

[9]  C. Haslett,et al.  Regulation of neutrophil apoptosis by tumor necrosis factor-alpha: requirement for TNFR55 and TNFR75 for induction of apoptosis in vitro. , 1997, Blood.

[10]  G. Turino,et al.  Atopy, Asthma, and Emphysema in Patients with Severe α -1-Antitrypysin Deficiency , 1997 .

[11]  J. Potempa,et al.  Biosynthesis of α1-Proteinase Inhibitor by Human Lung-derived Epithelial Cells* , 1997, The Journal of Biological Chemistry.

[12]  C. Haslett,et al.  Demonstration of reversible priming of human neutrophils using platelet-activating factor. , 1996, Blood.

[13]  J. Abrahams,et al.  Inhibitory conformation of the reactive loop of α1-antitrypsin , 1996, Nature Structural Biology.

[14]  P. Diaz,et al.  Alpha 1-antitrypsin deficiency: evaluation of bronchiectasis with CT. , 1996, Radiology.

[15]  C. Pusey,et al.  C-antineutrophil cytoplasmic antibody positivity in vasculitis patients is associated with the Z allele of alpha-1-antitrypsin, and P-antineutrophil cytoplasmic antibody positivity with the S allele. , 1996, Nephrology, dialysis, transplantation : official publication of the European Dialysis and Transplant Association - European Renal Association.

[16]  J. Whisstock,et al.  alpha 1-Antitrypsin Mmalton (Phe52-deleted) forms loop-sheet polymers in vivo. Evidence for the C sheet mechanism of polymerization. , 1995, The Journal of biological chemistry.

[17]  M. Lawson,et al.  Lack of effect of recombinant platelet-derived growth factor on human neutrophil function. , 1995, Journal of immunology.

[18]  D. Lomas,et al.  Mutations Which Impede Loop/Sheet Polymerization Enhance the Secretion of Human α1-Antitrypsin Deficiency Variants (*) , 1995, The Journal of Biological Chemistry.

[19]  D. Lomas,et al.  Preparation and Characterization of Latent α1-Antitrypsin (*) , 1995, The Journal of Biological Chemistry.

[20]  M. Adolphe,et al.  A non-isotopic, highly sensitive, fluorimetric, cell-cell adhesion microplate assay using calcein AM-labeled lymphocytes. , 1995, Journal of immunological methods.

[21]  D. Lomas,et al.  Alpha 1-antitrypsin Siiyama (Ser53-->Phe). Further evidence for intracellular loop-sheet polymerization. , 1993, The Journal of biological chemistry.

[22]  D. Lomas,et al.  Effect of the Z mutation on the physical and inhibitory properties of alpha 1-antitrypsin. , 1993, Biochemistry.

[23]  R. Senior,et al.  The serpin-enzyme complex (SEC) receptor mediates the neutrophil chemotactic effect of alpha-1 antitrypsin-elastase complexes and amyloid-beta peptide. , 1992, The Journal of clinical investigation.

[24]  D. Lomas,et al.  The mechanism of Z α1-antitrypsin accumulation in the liver , 1993, Nature.

[25]  B. Cronstein,et al.  Neuropeptides and inflammation. A somatostatin analog as a selective antagonist of neutrophil activation by substance P. , 1992, Arthritis and rheumatism.

[26]  R. Crystal,et al.  Neutrophil accumulation in the lung in alpha 1-antitrypsin deficiency. Spontaneous release of leukotriene B4 by alveolar macrophages. , 1991, The Journal of clinical investigation.

[27]  J. Travis,et al.  Acute phase protein stimulation by alpha 1-antichymotrypsin-cathepsin G complexes. Evidence for the involvement of interleukin-6. , 1990, The Journal of biological chemistry.

[28]  S. Zigmond Chemotactic response of neutrophils. , 1989, American journal of respiratory cell and molecular biology.

[29]  R. Senior,et al.  The inhibitory complex of human alpha 1-proteinase inhibitor and human leukocyte elastase is a neutrophil chemoattractant , 1988, The Journal of experimental medicine.

[30]  R. Stockley,et al.  Lung lavage fluid from patients with alpha 1-proteinase inhibitor deficiency or chronic obstructive bronchitis: anti-elastase function and cell profile. , 1987, Clinical science.

[31]  C. Nathan,et al.  Subcellular location and properties of bactericidal factors from human neutrophils , 1986, The Journal of experimental medicine.

[32]  R. Crystal,et al.  Expression of the alpha-1-antitrypsin gene in mononuclear phagocytes of normal and alpha-1-antitrypsin-deficient individuals. , 1986, The Journal of clinical investigation.

[33]  R. Crystal,et al.  Alpha 1-antitrypsin deficiency and emphysema caused by homozygous inheritance of non-expressing alpha 1-antitrypsin genes. , 1986, The New England journal of medicine.

[34]  J. Carlson,et al.  Risk of cirrhosis and primary liver cancer in alpha 1-antitrypsin deficiency. , 1986, The New England journal of medicine.

[35]  C. Haslett,et al.  Modulation of multiple neutrophil functions by preparative methods or trace concentrations of bacterial lipopolysaccharide. , 1985, The American journal of pathology.

[36]  R. Carrell,et al.  SMOKING, LUNG FUNCTION, AND α1-ANTITRYPSIN DEFICIENCY , 1985, The Lancet.

[37]  R. Huber,et al.  Human alpha 1-proteinase inhibitor. Crystal structure analysis of two crystal modifications, molecular model and preliminary analysis of the implications for function. , 1984, Journal of molecular biology.

[38]  R. Carrell,et al.  Structure and variation of human α1–antitrypsin , 1982, Nature.

[39]  W. Falk,et al.  Rapid quantitation of neutrophil chemotaxis: use of a polyvinylpyrrolidone-free polycarbonate membrane in a multiwell assembly. , 1980, Journal of immunological methods.

[40]  V. Ferrans,et al.  Inflammatory and immune processes in the human lung in health and disease: evaluation by bronchoalveolar lavage. , 1979, The American journal of pathology.

[41]  C. Larsson Natural history and life expectancy in severe alpha1-antitrypsin deficiency, Pi Z. , 2009, Acta medica Scandinavica.

[42]  T Sveger,et al.  Liver disease in alpha1-antitrypsin deficiency detected by screening of 200,000 infants. , 1976, The New England journal of medicine.

[43]  J. Jeppsson Amino acid substitution Glu→Lys in α1‐antitrypsin PiZ , 1976 .

[44]  A. Yoshida,et al.  Molecular abnormality of human alpha1-antitrypsin variant (Pi-ZZ) associated with plasma activity deficiency. , 1976, Proceedings of the National Academy of Sciences of the United States of America.

[45]  R. Simmons,et al.  Chemotaxis under agarose: a new and simple method for measuring chemotaxis and spontaneous migration of human polymorphonuclear leukocytes and monocytes. , 1975, Journal of immunology.

[46]  R. Talamo,et al.  α1-Antitrypsin Deficiency: A Variant with No Detectable α1-Antitrypsin , 1973, Science.

[47]  W. Krivit,et al.  Cirrhosis associated with alpha-1-antitrypsin deficiency: a previously unrecognized inherited disorder. , 1969, The Journal of laboratory and clinical medicine.

[48]  S. Eriksson Studies in alpha 1-antitrypsin deficiency. , 1965, Acta medica Scandinavica. Supplementum.

[49]  C. Laurell,et al.  The Electrophoretic α;1-Globulin Pattern of Serum in α;1-Antitrypsin Deficiency , 1963 .