Balasubramanyan N, Murphy A, O'Sullivan J, O'Connell E J
Department of Pediatric and Adolescent Medicine, Mayo Clinic, Rochester, MN 55905, USA.
Pediatr Pulmonol. 1997 Jan;23(1):55-61. doi: 10.1002/(sici)1099-0496(199701)23:1<55::aid-ppul7>3.0.co;2-o.
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.
我们描述了一名经活检确诊为间质性肺病（ILD）的男婴，他在口服皮质类固醇或环磷酰胺治疗无效后，对氯喹产生了反应。该男婴8日龄时出现呼吸窘迫和发绀。8周龄时的肺活检结果符合脱屑性间质性肺炎（DIP）。由于有两个兄弟姐妹在婴儿期死亡且尸检显示患有DIP的家族史，他在2周龄时接受了皮质类固醇治疗。8.5个月时，由于对皮质类固醇治疗无反应，我们的患者接受了环磷酰胺治疗。14个月大时，他开始在使用皮质类固醇的基础上加用氯喹治疗，并在3周内出现显著反应。自开始这种治疗以来，该患者仅使用氯喹持续治疗已成功维持了9年多。
