Literature DB >> 7028785

Replacement therapy of alpha 1-antitrypsin deficiency. Reversal of protease-antiprotease imbalance within the alveolar structures of PiZ subjects.

J E Gadek, H G Klein, P V Holland, R G Crystal.   

Abstract

The emphysema associated with the inherited serum deficiency of alpha 1-antitrypsin appears to result from an imbalance between neutrophil elastase and its major inhibitor within the alveolar structures. In the present study we assessed the feasibility of reversing this biochemical defect within the lung via parenteral replacement therapy with an alpha 1-antitrypsin concentrate of normal plasma. A 20--40% polyethylene glycol precipitate of pooled human donor plasma was used to obtain an enriched alpha 1-antitrypsin concentrate devoid of hepatitis B antigen and immunoglobulins. Using this material, five individuals with severe serum alpha 1-antitrypsin deficiency (PiZ phenotype) and advanced emphysema received 4 g of alpha 1-antitrypsin intravenously at weekly intervals for four doses. During this period of weekly replacement therapy alpha 1-antitrypsin serum levels were maintained at greater than or equal to 70 mg/dl, the level likely required for effective antielastase protection of the lung. In addition, assessment of lower respiratory tract antielastase activity by bronchoalveolar lavage demonstrated that parenteral replacement of alpha 1-antitrypsin resulted in establishment of effective antielastase activity within the alveolar structures. There were no untoward side effects consequent to this approach to the replacement therapy of alpha 1-antitrypsin. These results demonstrate that the parenteral replacement of alpha 1-antitrypsin provides a means of obtaining elastase-antielastase balance within the lung of individuals with this serum protease inhibitor deficiency.

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Year:  1981        PMID: 7028785      PMCID: PMC370909          DOI: 10.1172/jci110360

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


  29 in total

1.  Purification and preliminary characterization of human leukocyte elastasel.

Authors:  J C Taylor; I P Crawford
Journal:  Arch Biochem Biophys       Date:  1975-07       Impact factor: 4.013

2.  Obstructive lung disease in heterozygotes for alpha-1 antitrypsin deficiency.

Authors:  F Kueppers; A Dönhardt
Journal:  Ann Intern Med       Date:  1974-02       Impact factor: 25.391

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Authors:  G Mancini; A O Carbonara; J F Heremans
Journal:  Immunochemistry       Date:  1965-09

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Journal:  Thromb Diath Haemorrh Suppl       Date:  1968

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

6.  A new method for determination of alpha-1-antitrypsin phenotypes using isoelectric focusing on polyacrylamide gel slabs.

Authors:  R C Allen; R A Harley; R C Talamo
Journal:  Am J Clin Pathol       Date:  1974-12       Impact factor: 2.493

Review 7.  Alpha1-antitrypsin and its deficiency.

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

8.  Distribution and elimination of exogenous alpha1-antitrypsin.

Authors:  S Makino; C E Reed
Journal:  J Lab Clin Med       Date:  1970-05

9.  Determinants of chronic obstructive pulmonary disease in patients with intermediate levels of alpha-antitrypsin.

Authors:  R Klayton; R Fallat; A B Cohen
Journal:  Am Rev Respir Dis       Date:  1975-07

10.  Protease inhibitors in patients with chronic obstructive pulmonary disease: the alpha-antitrypsin heterozygote controversy.

Authors:  D W Cox; V H Hoeppner; H Levison
Journal:  Am Rev Respir Dis       Date:  1976-05
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  60 in total

Review 1.  Alpha-1-antitrypsin deficiency: what next?

Authors:  R A Stockley
Journal:  Thorax       Date:  2000-07       Impact factor: 9.139

Review 2.  Molecular biology and respiratory disease. 7. The alpha 1 antitrypsin gene and chronic lung disease.

Authors:  N Kalsheker; K Morgan
Journal:  Thorax       Date:  1990-10       Impact factor: 9.139

Review 3.  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

Review 4.  alpha-1-Antitrypsin and the pathogenesis of emphysema.

Authors:  R A Stockley
Journal:  Lung       Date:  1987       Impact factor: 2.584

5.  Biochemical reaction of alpha 1 antitrypsin during the substitution therapy of patients with homozygote PI-ZZ deficit.

Authors:  A Gillissen; E W Schmidt; B Rasche; W T Ulmer
Journal:  Klin Wochenschr       Date:  1989-03-15

6.  Alpha 1-antitrypsin--an investigative odyssey from the benchtop to the bedside.

Authors:  J E Gadek
Journal:  West J Med       Date:  1987-07

Review 7.  Gene Therapy for Alpha-1 Antitrypsin Deficiency Lung Disease.

Authors:  Maria J Chiuchiolo; Ronald G Crystal
Journal:  Ann Am Thorac Soc       Date:  2016-08

Review 8.  Chronic obstructive pulmonary disease: less common causes--an algorithm for the primary care physician.

Authors:  R C St John; J E Gadek; E R Pacht
Journal:  J Gen Intern Med       Date:  1993-10       Impact factor: 5.128

9.  Safety and efficacy of alpha-1-antitrypsin augmentation therapy in the treatment of patients with alpha-1-antitrypsin deficiency.

Authors:  Irina Petrache; Joud Hajjar; Michael Campos
Journal:  Biologics       Date:  2009-07-13

Review 10.  Targeted treatment in COPD: a multi-system approach for a multi-system disease.

Authors:  David Anderson; William Macnee
Journal:  Int J Chron Obstruct Pulmon Dis       Date:  2009-09-01
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