Literature DB >> 3264124

Clinical features and history of the destructive lung disease associated with alpha-1-antitrypsin deficiency of adults with pulmonary symptoms.

M L Brantly1, L D Paul, B H Miller, R T Falk, M Wu, R G Crystal.   

Abstract

Alpha-1-antitrypsin (alpha 1AT) deficiency is a hereditary disorder characterized in adults by a high risk for the development of severe destructive lung disease at an early age. The present study was designed to draw conclusions concerning the characteristics of a referral population of 124 patients with alpha 1AT deficiency and symptomatic emphysema. Typically, the alpha 1AT level was 30 mg/dl, and the alpha 1AT phenotype was almost always PiZZ. The individuals in this population were most often male, caucasian, and ex-smokers, and they had become dyspneic between 25 and 40 yr of age. Most routine blood tests were normal. The chest radiographs and ventilation-perfusion studies typically showed abnormalities with a lower zone distribution, and about one third of the study population had evidence suggestive of pulmonary hypertension. Lung function tests were typical for emphysema; the FEV1 and DLCO were the parameters most dramatically reduced, and the annual rate of decline of those parameters was greater than that of the general population. The cumulative probability of survival of this population indicated a significantly shortened lifespan with a mean survival of 16% at 60 yr of age compared with 85% for normal persons.

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Year:  1988        PMID: 3264124     DOI: 10.1164/ajrccm/138.2.327

Source DB:  PubMed          Journal:  Am Rev Respir Dis        ISSN: 0003-0805


  63 in total

Review 1.  Factors influencing airway inflammation in chronic obstructive pulmonary disease.

Authors:  A Hill; S Gompertz; R Stockley
Journal:  Thorax       Date:  2000-11       Impact factor: 9.139

Review 2.  Alpha-1 antitrypsin deficiency.

Authors:  R A Primhak; M S Tanner
Journal:  Arch Dis Child       Date:  2001-07       Impact factor: 3.791

3.  Characteristics of Alpha-1 Antitrypsin-Deficient Individuals in the Long-term Oxygen Treatment Trial and Comparison with Other Subjects with Chronic Obstructive Pulmonary Disease.

Authors:  James K Stoller; Loutfi S Aboussouan; Richard E Kanner; Laura A Wilson; Phil Diaz; Robert Wise
Journal:  Ann Am Thorac Soc       Date:  2015-12

Review 4.  Airway hydration and COPD.

Authors:  Arunava Ghosh; R C Boucher; Robert Tarran
Journal:  Cell Mol Life Sci       Date:  2015-06-12       Impact factor: 9.261

Review 5.  Natural history of emphysema.

Authors:  Omar A Minai; Joshua Benditt; Fernando J Martinez
Journal:  Proc Am Thorac Soc       Date:  2008-05-01

Review 6.  Gene Therapy for Alpha-1 Antitrypsin Deficiency Lung Disease.

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

7.  Survival in severe alpha-1-antitrypsin deficiency (PiZZ).

Authors:  Hanan A Tanash; Peter M Nilsson; Jan-Ake Nilsson; Eeva Piitulainen
Journal:  Respir Res       Date:  2010-04-26

8.  Sputum chemotactic activity in chronic obstructive pulmonary disease: effect of alpha(1)-antitrypsin deficiency and the role of leukotriene B(4) and interleukin 8.

Authors:  I S Woolhouse; D L Bayley; R A Stockley
Journal:  Thorax       Date:  2002-08       Impact factor: 9.139

9.  Risk factors for symptom onset in PI*Z alpha-1 antitrypsin deficiency.

Authors:  Annyce S Mayer; James K Stoller; Sverre Vedal; A James Ruttenber; Matt Strand; Robert A Sandhaus; Lee S Newman
Journal:  Int J Chron Obstruct Pulmon Dis       Date:  2006

10.  Alpha-1-antitrypsin phenotypes in adult liver disease patients.

Authors:  Aleksandra Topic; Tamara Alempijevic; Aleksandra Sokic Milutinovic; Nada Kovacevic
Journal:  Ups J Med Sci       Date:  2009       Impact factor: 2.384

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