Familial interstitial lung disease in children: response to chloroquine treatment in one sibling with desquamative interstitial pneumonitis.
Identifieur interne : 001172 ( Ncbi/Curation ); précédent : 001171; suivant : 001173Familial interstitial lung disease in children: response to chloroquine treatment in one sibling with desquamative interstitial pneumonitis.
Auteurs : N. Balasubramanyan [États-Unis] ; A. Murphy ; J. O'Sullivan ; E J O'ConnellSource :
- Pediatric pulmonology [ 8755-6863 ] ; 1997.
Descripteurs français
- KwdFr :
- MESH :
- génétique : Pneumopathies interstitielles.
- traitement médicamenteux : Pneumopathies interstitielles.
- usage thérapeutique : Chloroquine.
- Humains, Mâle, Nouveau-né.
English descriptors
- KwdEn :
- MESH :
- chemical , therapeutic use : Chloroquine.
- drug therapy : Lung Diseases, Interstitial.
- genetics : Lung Diseases, Interstitial.
- Humans, Infant, Newborn, Male.
Abstract
We describe a male infant with biopsy-confirmed interstitial lung disease (ILD) who responded to chloroquine, after he failed to improve on oral corticosteroids or cyclophosphamide. The infant presented at 8 days of age with respiratory distress and cyanosis. Lung biopsy at 8 weeks of age was consistent with desquamative interstitial pneumonitis (DIP). He was treated with corticosteroids at 2 weeks of age because of a family history of two siblings who died during infancy and who had DIP on postmortem examination. At 8.5 months, our patient was treated with cyclophosphamide because of lack of response to corticosteroids therapy. At 14 months of age, he began treatment with chloroquine in addition to corticosteroids and had a dramatic response within 3 weeks. The patient has been maintained successfully on continuous treatment with chloroquine alone for more than 9 years since this treatment was started.
DOI: 10.1002/(sici)1099-0496(199701)23:1<55::aid-ppul7>3.0.co;2-o
PubMed: 9035199
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pubmed:9035199Le document en format XML
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Balasubramanyan</name><affiliation wicri:level="2"><nlm:affiliation>Department of Pediatric and Adolescent Medicine, Mayo Clinic, Rochester, MN 55905, USA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Pediatric and Adolescent Medicine, Mayo Clinic, Rochester, MN 55905</wicri:regionArea><placeName><region type="state">Minnesota</region></placeName></affiliation></author><author><name sortKey="Murphy, A" sort="Murphy, A" uniqKey="Murphy A" first="A" last="Murphy">A. Murphy</name></author><author><name sortKey="O Sullivan, J" sort="O Sullivan, J" uniqKey="O Sullivan J" first="J" last="O'Sullivan">J. O'Sullivan</name></author><author><name sortKey="O Connell, E J" sort="O Connell, E J" uniqKey="O Connell E" first="E J" last="O'Connell">E J O'Connell</name></author></analytic><series><title level="j">Pediatric pulmonology</title><idno type="ISSN">8755-6863</idno><imprint><date when="1997" type="published">1997</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Chloroquine (therapeutic use)</term><term>Humans</term><term>Infant, Newborn</term><term>Lung Diseases, Interstitial (drug therapy)</term><term>Lung Diseases, Interstitial (genetics)</term><term>Male</term></keywords><keywords scheme="KwdFr" xml:lang="fr"><term>Chloroquine (usage thérapeutique)</term><term>Humains</term><term>Mâle</term><term>Nouveau-né</term><term>Pneumopathies interstitielles (génétique)</term><term>Pneumopathies interstitielles (traitement médicamenteux)</term></keywords><keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>Chloroquine</term></keywords><keywords scheme="MESH" qualifier="drug therapy" xml:lang="en"><term>Lung Diseases, Interstitial</term></keywords><keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Lung Diseases, Interstitial</term></keywords><keywords scheme="MESH" qualifier="génétique" xml:lang="fr"><term>Pneumopathies interstitielles</term></keywords><keywords scheme="MESH" qualifier="traitement médicamenteux" xml:lang="fr"><term>Pneumopathies interstitielles</term></keywords><keywords scheme="MESH" qualifier="usage thérapeutique" xml:lang="fr"><term>Chloroquine</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Humans</term><term>Infant, Newborn</term><term>Male</term></keywords><keywords scheme="MESH" xml:lang="fr"><term>Humains</term><term>Mâle</term><term>Nouveau-né</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">We describe a male infant with biopsy-confirmed interstitial lung disease (ILD) who responded to chloroquine, after he failed to improve on oral corticosteroids or cyclophosphamide. The infant presented at 8 days of age with respiratory distress and cyanosis. Lung biopsy at 8 weeks of age was consistent with desquamative interstitial pneumonitis (DIP). He was treated with corticosteroids at 2 weeks of age because of a family history of two siblings who died during infancy and who had DIP on postmortem examination. At 8.5 months, our patient was treated with cyclophosphamide because of lack of response to corticosteroids therapy. At 14 months of age, he began treatment with chloroquine in addition to corticosteroids and had a dramatic response within 3 weeks. The patient has been maintained successfully on continuous treatment with chloroquine alone for more than 9 years since this treatment was started.</div></front></TEI></record>Pour manipuler ce document sous Unix (Dilib)
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