Document Detail


Clinical features, long-term follow-up and outcome of a large cohort of patients with Chronic Granulomatous Disease: an Italian multicenter study.
MedLine Citation:
PMID:  18037347     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
A retrospective clinical and immunological survey was conducted in 60 patients with Chronic Granulomatous Disease. A prospective controlled non-randomized study of the efficacy of long-term IFNgamma treatment was carried out. The mean age at the time of diagnosis was 4.4 years; mean duration of follow-up was 10.4 years. Lung and skin infections were the most frequent manifestations both prior to diagnosis and during follow-up. Aspergillus species was the first cause of infection and of death in our cohort. The mortality rate was 13%. Long term prophylaxis with IFNgamma did not significantly change the rate of total infection per patient-year compared to controls (p=0.07). Our data provide clear evidence that protocols of continuing intensive surveillance and monitoring of compliance with anti-infective regimens may significantly improve the quality of life and long-term survival in patients with CGD. No evidence justifying long-term prophylaxis with IFNgamma was obtained.
Authors:
Baldassarre Martire; Roberto Rondelli; Annarosa Soresina; Claudio Pignata; Teresa Broccoletti; Andrea Finocchi; Paolo Rossi; Marco Gattorno; Marco Rabusin; Chiara Azzari; Rosa M Dellepiane; Maria C Pietrogrande; Antonino Trizzino; Paolo Di Bartolomeo; Silvana Martino; Luigi Carpino; Fausto Cossu; Franco Locatelli; Rita Maccario; Paolo Pierani; Maria C Putti; Achille Stabile; Luigi D Notarangelo; Alberto G Ugazio; Alessandro Plebani; Domenico De Mattia;
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Publication Detail:
Type:  Journal Article; Multicenter Study     Date:  2007-11-26
Journal Detail:
Title:  Clinical immunology (Orlando, Fla.)     Volume:  126     ISSN:  1521-6616     ISO Abbreviation:  Clin. Immunol.     Publication Date:  2008 Feb 
Date Detail:
Created Date:  2008-01-14     Completed Date:  2008-02-28     Revised Date:  2008-11-21    
Medline Journal Info:
Nlm Unique ID:  100883537     Medline TA:  Clin Immunol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  155-64     Citation Subset:  IM    
Affiliation:
Dipartimento di Biomedicina dell'Età Evolutiva, Università di Bari, Italy. baldo.martire@bioetaev.uniba.it
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MeSH Terms
Descriptor/Qualifier:
Anti-Infective Agents / therapeutic use
Antifungal Agents / therapeutic use
Antiviral Agents / therapeutic use
Bacterial Infections / etiology*,  prevention & control
Child
Child, Preschool
Cohort Studies
Female
Follow-Up Studies
Granulomatous Disease, Chronic* / complications,  diagnosis,  mortality
Humans
Infant
Interferon-gamma / therapeutic use
Italy
Itraconazole / therapeutic use
Kaplan-Meiers Estimate
Male
Retrospective Studies
Treatment Outcome
Trimethoprim-Sulfamethoxazole Combination / therapeutic use
Chemical
Reg. No./Substance:
0/Anti-Infective Agents; 0/Antifungal Agents; 0/Antiviral Agents; 8064-90-2/Trimethoprim-Sulfamethoxazole Combination; 82115-62-6/Interferon-gamma; 84625-61-6/Itraconazole

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


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