Document Detail


Reversible airway obstruction in children with ataxia telangiectasia.
MedLine Citation:
PMID:  20146367     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
INTRODUCTION: Lung disease is a significant cause of the short life span of ataxia telangiectasia (A-T) patients. Objective lung function measurements are difficult to achieve in A-T. AIM: To assess lung function by spirometry in relation to the clinical characteristics of A-T patients followed up at the Israeli Ataxia Telangiectasia National Clinic. PATIENTS AND METHODS: Medical and spirometry data were collected from 27 A-T patients during 2004-2007. Laboratory, nutritional condition, mode of treatment, pulmonary status, and malignancies were assessed. The spirometry values FVC, FEV(1), FEV(0.5), FEF(25-75), PEF and time rise to peak flow were analyzed individually and values were compared to those of healthy age-matched children. RESULTS: Eleven patients (40.7%) were found to suffer from asthma according to clinical symptoms and response to bronchodilators. We found significant reduction in FEV(1) and FEV(0.5) (z-scores: -0.84 + or - 0.7 SD, -0.7 + or - 0.6 SD; P = 0.0014 and P = 0.003, respectively), in relation to healthy predicted values. FEF(25-75) was significantly lower than that in healthy children in 5 of 11 asthmatic patients. All 27 patients showed higher than healthy FEV(1)/FVC and FEV(0.5)/FVC ratios (z-scores 0.68 + or - 0.99 SD, P < 0.0015, and 2.12 + or - 1.50 SD, P < 0.0015, respectively). The rise time to peak flow was three-fold longer than that of healthy children. CONCLUSION: Obstructive lung disease is common among A-T patients. Maximal peak flow reduction and prolonged rise time to peak flow may be the first signs of pulmonary involvement in these patients. Early treatment with anti-asthma therapy, bronchodilators, and steroids, may prevent further pulmonary deterioration and improve the prognosis of A-T patients.
Authors:
Yackov Berkun; Daphna Vilozni; Yonit Levi; Sheron Borik; Dalia Waldman; Raz Somech; Andreea Nissenkorn; Ori Efrati
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Pediatric pulmonology     Volume:  45     ISSN:  1099-0496     ISO Abbreviation:  Pediatr. Pulmonol.     Publication Date:  2010 Mar 
Date Detail:
Created Date:  2010-02-26     Completed Date:  2010-06-16     Revised Date:  2010-09-30    
Medline Journal Info:
Nlm Unique ID:  8510590     Medline TA:  Pediatr Pulmonol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  230-5     Citation Subset:  IM    
Affiliation:
Ataxia Telangiectasia National Clinic, Chaim Sheba Medical Center, Tel Hashomer, affiliated with the Sackler Medical School, Tel-Aviv University, Israel.
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MeSH Terms
Descriptor/Qualifier:
Administration, Inhalation
Adolescent
Anti-Asthmatic Agents / administration & dosage
Asthma / complications,  drug therapy
Ataxia Telangiectasia / complications*
Bronchodilator Agents / administration & dosage*
Child
Child, Preschool
Female
Forced Expiratory Volume
Humans
Male
Pulmonary Disease, Chronic Obstructive / drug therapy*,  etiology*,  physiopathology
Spirometry
Vital Capacity
Young Adult
Chemical
Reg. No./Substance:
0/Anti-Asthmatic Agents; 0/Bronchodilator Agents
Comments/Corrections
Comment In:
Pediatr Pulmonol. 2010 Oct;45(10):1043-4; author reply 1045   [PMID:  20597085 ]

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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