Literature DB >> 12149531

Respiratory symptoms and lung function in young adults with severe alpha(1)-antitrypsin deficiency (PiZZ).

E Piitulainen1, T Sveger.   

Abstract

BACKGROUND: Neonatal screening for alpha(1)-antitrypsin (AAT) deficiency was undertaken in Sweden between 1972 and 1974 when 129 infants with severe AAT deficiency (phenotype PiZ) were identified. The cohort has been followed up prospectively.
METHODS: 124 PiZ subjects, still alive and still living in Sweden, were invited to a follow up examination at about 22 years of age. The check up included a clinical examination, spirometric tests, and a questionnaire on smoking habits and respiratory symptoms.
RESULTS: Ninety eight subjects (97 PiZZ and 1 PiZ-) subjects attended the follow up. The mean age of the subjects was 22.5 years (range 19.8-24.8). The mean (SD) forced expiratory volume in 1 second (FEV(1)) was 98 (14)% predicted, vital capacity (VC) was 103 (14)% predicted, and the mean FEV(1)/VC ratio was 83 (7)%. Eighty six subjects had previously undergone spirometric tests. The median follow up time was 4.3 years (range 0.9-7.3). The mean annual change in FEV(1) (% predicted) was -1.2% (95% CI -2.1 to -0.3), in VC (% predicted) was -1.5% (95% CI -2.0 to -0.9), and in the FEV(1)/VC ratio (%) was -0.3% (95% CI -0.7 to 0.2). Twenty eight individuals (29%) reported recurrent wheezing. Fifteen subjects (15%) had been diagnosed by a physician as having asthma. Eighteen subjects reported that they had smoked at some time; 10 were current smokers. The mean number of pack years among the ever smokers was 3.4 (range 0.6-10.5). Ten of 18 ever-smokers and 18 of 80 non-smokers reported recurrent wheezing (p<0.01), while exertional dyspnoea was reported by six ever smokers and 11 non-smokers (p<0.05). Lung function test results did not differ significantly between ever smokers and non-smokers.
CONCLUSIONS: Young PiZ adults have essentially normal lung function, but have a high prevalence of asthma symptoms. Smoking in these individuals is associated with an increased frequency of respiratory symptoms.

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Year:  2002        PMID: 12149531      PMCID: PMC1746413          DOI: 10.1136/thorax.57.8.705

Source DB:  PubMed          Journal:  Thorax        ISSN: 0040-6376            Impact factor:   9.139


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9.  Lung function in adolescents with alpha 1-antitrypsin deficiency.

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3.  Determinants of airflow obstruction in severe alpha-1-antitrypsin deficiency.

Authors:  Dawn L Demeo; Robert A Sandhaus; Alan F Barker; Mark L Brantly; Edward Eden; N Gerard McElvaney; Stephen Rennard; Esteban Burchard; James M Stocks; James K Stoller; Charlie Strange; Gerard M Turino; Edward J Campbell; Edwin K Silverman
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Review 7.  Alpha-1 antitrypsin deficiency: a conformational disease associated with lung and liver manifestations.

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8.  Health status and lung function in the Swedish alpha 1-antitrypsin deficient cohort, identified by neonatal screening, at the age of 37-40 years.

Authors:  Eeva Piitulainen; Behrouz Mostafavi; Hanan A Tanash
Journal:  Int J Chron Obstruct Pulmon Dis       Date:  2017-02-02

Review 9.  Update on α1-antitrypsin deficiency.

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10.  Environmental, occupational, and genetic risk factors for alpha-1 antitrypsin deficiency.

Authors:  Richard R Sharp; Frederick de Serres; Lee Newman; Robert A Sandhaus; John W Walsh; Ernie Hood; G Jean Harry
Journal:  Environ Health Perspect       Date:  2003-11       Impact factor: 9.031

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