Literature DB >> 19926349

Pulmonary outcome differences in U.S. and French cystic fibrosis cohorts diagnosed through newborn screening.

Aimee C Walsh1, Gilles Rault, Zhanhai Li, Virginie Scotet, Ingrid Duguépéroux, Claude Férec, Michel Roussey, Anita Laxova, Philip M Farrell.   

Abstract

BACKGROUND: A comparison of the longitudinal progression of lung disease in cystic fibrosis patients identified through newborn screening (NBS) in cohorts located in two different countries has never been performed and was the primary objective of this study.
METHODS: The study included 56 patients in Brittany diagnosed through NBS between 1989 and 1994 and 69 similar patients in Wisconsin between 1985 and 1994. The onset and progression of lung disease was radiographically quantified using the Wisconsin Chest X-ray (WCXR) scoring system. A single pediatric pulmonologist blinded to all identifiers scored the films.
RESULTS: Generalized estimating equation analyses adjusted for age, genotype, sex, pancreatic insufficiency, and meconium ileus showed worse WCXR scores in Brittany patients compared to Wisconsin patients (average score difference=4.48; p<0.001). Percent predicted FEV1 was also worse among Brittany patients (p<0.001).
CONCLUSIONS: The finding of milder radiographically-quantified lung disease using the WCXR scoring system, as well as better FEV1 values, may be explained by variations in nutrition, environmental exposures, or healthcare delivery.

Entities:  

Mesh:

Year:  2009        PMID: 19926349      PMCID: PMC2818431          DOI: 10.1016/j.jcf.2009.10.004

Source DB:  PubMed          Journal:  J Cyst Fibros        ISSN: 1569-1993            Impact factor:   5.482


  29 in total

1.  Evidence for spread of a clonal strain of Pseudomonas aeruginosa among cystic fibrosis clinics.

Authors:  David Armstrong; Scott Bell; Michael Robinson; Peter Bye; Barbara Rose; Colin Harbour; Crystal Lee; Helen Service; Michael Nissen; Melanie Syrmis; Claire Wainwright
Journal:  J Clin Microbiol       Date:  2003-05       Impact factor: 5.948

2.  Comparing the clinical evolution of cystic fibrosis screened neonatally to that of cystic fibrosis diagnosed from clinical symptoms: a 10-year retrospective study in a French region (Brittany).

Authors:  D Siret; G Bretaudeau; B Branger; A Dabadie; M Dagorne; V David; M de Braekeleer; V Moisan-Petit; G Picherot; G Rault; V Storni; M Roussey
Journal:  Pediatr Pulmonol       Date:  2003-05

3.  Complete and rapid scanning of the cystic fibrosis transmembrane conductance regulator (CFTR) gene by denaturing high-performance liquid chromatography (D-HPLC): major implications for genetic counselling.

Authors:  C Le Maréchal; M P Audrézet; I Quéré; O Raguénès; S Langonné; C Férec
Journal:  Hum Genet       Date:  2001-04       Impact factor: 4.132

4.  Early diagnosis of cystic fibrosis through neonatal screening prevents severe malnutrition and improves long-term growth. Wisconsin Cystic Fibrosis Neonatal Screening Study Group.

Authors:  P M Farrell; M R Kosorok; M J Rock; A Laxova; L Zeng; H C Lai; G Hoffman; R H Laessig; M L Splaingard
Journal:  Pediatrics       Date:  2001-01       Impact factor: 7.124

5.  Spectrum of mutations in the CFTR gene of patients with classical and atypical forms of cystic fibrosis from southwestern Sweden: identification of 12 novel mutations.

Authors:  B Strandvik; E Björck; M Fallström; E Gronowitz; J Thountzouris; A Lindblad; D Markiewicz; J Wahlström; L C Tsui; J Zielenski
Journal:  Genet Test       Date:  2001

6.  Longitudinal evaluation of bronchopulmonary disease in children with cystic fibrosis.

Authors:  Philip M Farrell; Zhanhai Li; Michael R Kosorok; Anita Laxova; Christopher G Green; Jannette Collins; Hui-Chuan Lai; Linda M Makholm; Michael J Rock; Mark L Splaingard
Journal:  Pediatr Pulmonol       Date:  2003-09

7.  Association between initial disease presentation, lung disease outcomes, and survival in patients with cystic fibrosis.

Authors:  HuiChuan J Lai; Yu Cheng; Hyungjun Cho; Michael R Kosorok; Philip M Farrell
Journal:  Am J Epidemiol       Date:  2004-03-15       Impact factor: 4.897

8.  Longitudinal changes in growth parameters are correlated with changes in pulmonary function in children with cystic fibrosis.

Authors:  Michelle L Peterson; David R Jacobs; Carlos E Milla
Journal:  Pediatrics       Date:  2003-09       Impact factor: 7.124

9.  Correlation of six different cystic fibrosis chest radiograph scoring systems with clinical parameters.

Authors:  Suzanne Terheggen-Lagro; Neeltje Truijens; Noor van Poppel; Vincent Gulmans; Johan van der Laag; Cornelis van der Ent
Journal:  Pediatr Pulmonol       Date:  2003-06

10.  Detection of a widespread clone of Pseudomonas aeruginosa in a pediatric cystic fibrosis clinic.

Authors:  David S Armstrong; Gillian M Nixon; Rosemary Carzino; Andrea Bigham; John B Carlin; Roy M Robins-Browne; Keith Grimwood
Journal:  Am J Respir Crit Care Med       Date:  2002-10-01       Impact factor: 21.405

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  2 in total

1.  Rate of improvement of CF life expectancy exceeds that of general population--observational death registration study.

Authors:  Matthew N Hurley; Tricia M McKeever; Andrew P Prayle; Andrew W Fogarty; Alan R Smyth
Journal:  J Cyst Fibros       Date:  2014-01-10       Impact factor: 5.482

2.  Showing Value in Newborn Screening: Challenges in Quantifying the Effectiveness and Cost-Effectiveness of Early Detection of Phenylketonuria and Cystic Fibrosis.

Authors:  Scott D Grosse
Journal:  Healthcare (Basel)       Date:  2015-11-11
  2 in total

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