Ten‐year follow up of hydroxychloroquine treatment for ABCA3 deficiency
Identifieur interne : 001048 ( Main/Exploration ); précédent : 001047; suivant : 001049Ten‐year follow up of hydroxychloroquine treatment for ABCA3 deficiency
Auteurs : Michael Williamson [Royaume-Uni] ; Colin Wallis [Royaume-Uni]Source :
- Pediatric Pulmonology [ 8755-6863 ] ; 2014-03.
Descripteurs français
- KwdFr :
- MESH :
- déficit : Transporteurs ABC.
- traitement médicamenteux : Pneumopathies interstitielles.
- usage thérapeutique : Hydroxychloroquine, Immunosuppresseurs.
- Adolescent, Enfant, Enfant d'âge préscolaire, Femelle, Humains, Résultat thérapeutique, Études longitudinales.
English descriptors
- KwdEn :
- MESH :
- chemical , deficiency : ATP-Binding Cassette Transporters.
- chemical , therapeutic use : Hydroxychloroquine, Immunosuppressive Agents.
- drug therapy : Lung Diseases, Interstitial.
- Adolescent, Child, Child, Preschool, Female, Humans, Longitudinal Studies, Treatment Outcome.
Abstract
We present a child with interstitial lung disease (ILD) who was diagnosed with desquamative interstitial pneumonitis following CT thorax and open lung biopsy aged 2 years. Subsequently, surfactant protein gene analysis revealed mutations in the ABCA3 gene. This case of ABCA3 deficient ILD describes a stable clinical course over 10 years and highlights the potential benefit of hydroxychloroquine for this condition. Pediatr Pulmonol. 2014; 49:299–301. © 2013 Wiley Periodicals, Inc.
Url:
DOI: 10.1002/ppul.22811
Affiliations:
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Le document en format XML
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<front><div type="abstract" xml:lang="en">We present a child with interstitial lung disease (ILD) who was diagnosed with desquamative interstitial pneumonitis following CT thorax and open lung biopsy aged 2 years. Subsequently, surfactant protein gene analysis revealed mutations in the ABCA3 gene. This case of ABCA3 deficient ILD describes a stable clinical course over 10 years and highlights the potential benefit of hydroxychloroquine for this condition. Pediatr Pulmonol. 2014; 49:299–301. © 2013 Wiley Periodicals, Inc.</div>
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