Successful treatment of idiopathic pulmonary capillaritis with intravenous cyclophosphamide

Pediatric Pulmonology
Frances FlanaganDubhfeasa M Slattery

Abstract

Idiopathic pulmonary hemosiderosis (IPH), a subtype of diffuse alveolar hemorrhage is a rare condition, first described by Virchow in 1864. Historically, it manifests in children in the first decade of life with the combination of hemoptysis, iron deficiency anemia, and alveolar infiltrates on chest radiograph. More recently, diffuse alveolar hemorrhage has been classified by the absence or presence of pulmonary capillaritis (PC), the latter carrying a potential for a poorer outcome. While systemic corticosteroids remain the first line treatment option, other immune modulators have been trailed including hydroxychloroquine, azathioprine, 6-mercaptopurine, and cyclophosphamide with varying results. Our case demonstrates for the first time, the successful use of intravenous cyclophosphamide in the management of chronic idiopathic PC.

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Citations

Apr 28, 2016·Pediatric Pulmonology·Mustafa ErkoçoğluCan Naci Kocabaş
Oct 4, 2018·The Journal of International Medical Research·Yajun ZhangYuhong Tao
Sep 21, 2016·Italian Journal of Pediatrics·Luca CastellazziSusanna Esposito
Dec 20, 2016·Biomédica : revista del Instituto Nacional de Salud·Ana Madeleine Barrera, Leslie Vargas
Nov 28, 2020·Respiratory Medicine·Biplab K Saha
Mar 12, 2021·Journal of Paediatrics and Child Health·Susan TelencoeJonathan H Rayment

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