Ten-year follow up of hydroxychloroquine treatment for ABCA3 deficiency.
Identifieur interne : 000215 ( Ncbi/Merge ); précédent : 000214; suivant : 000216Ten-year follow up of hydroxychloroquine treatment for ABCA3 deficiency.
Auteurs : Michael Williamson [Royaume-Uni] ; Colin WallisSource :
- Pediatric pulmonology [ 1099-0496 ] ; 2014.
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.
DOI: 10.1002/ppul.22811
PubMed: 24115460
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pubmed:24115460Le document en format XML
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<term>Female</term>
<term>Humans</term>
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<term>Hydroxychloroquine (usage thérapeutique)</term>
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<term>Résultat thérapeutique</term>
<term>Transporteurs ABC (déficit)</term>
<term>Études longitudinales</term>
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<term>Immunosuppressive Agents</term>
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<keywords scheme="MESH" xml:lang="fr"><term>Adolescent</term>
<term>Enfant</term>
<term>Enfant d'âge préscolaire</term>
<term>Femelle</term>
<term>Humains</term>
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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.</div>
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<Issue>3</Issue>
<PubDate><Year>2014</Year>
<Month>Mar</Month>
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<Title>Pediatric pulmonology</Title>
<ISOAbbreviation>Pediatr. Pulmonol.</ISOAbbreviation>
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<ArticleTitle>Ten-year follow up of hydroxychloroquine treatment for ABCA3 deficiency.</ArticleTitle>
<Pagination><MedlinePgn>299-301</MedlinePgn>
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<Abstract><AbstractText>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.</AbstractText>
<CopyrightInformation>© 2013 Wiley Periodicals, Inc.</CopyrightInformation>
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<ForeName>Michael</ForeName>
<Initials>M</Initials>
<AffiliationInfo><Affiliation>Great Ormond Street Hospital Respiratory Department, Great Ormond Street Hospital, London, WC1N 3JH, UK.</Affiliation>
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<ChemicalList><Chemical><RegistryNumber>0</RegistryNumber>
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