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Familial interstitial lung disease in children: Response to chloroquine treatment in one sibling with desquamative interstitial pneumonitis
Author(s) -
Balasubramanyan Napa,
Murphy Anne,
O'Sullivan Joe,
O'Connell Edward J.
Publication year - 1997
Publication title -
pediatric pulmonology
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 0.866
H-Index - 106
eISSN - 1099-0496
pISSN - 8755-6863
DOI - 10.1002/(sici)1099-0496(199701)23:1<55::aid-ppul7>3.0.co;2-o
Subject(s) - medicine , chloroquine , interstitial lung disease , respiratory distress , cyclophosphamide , lung biopsy , pneumonitis , sibling , lung , respiratory disease , surgery , pediatrics , chemotherapy , pathology , psychology , developmental psychology , malaria
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. Pediatr. Pulmonol. 1997; 23:55–61. © 1997 Wiley‐Liss, Inc.