Premium
Hughes Stovin: Sustained remission and regression of pulmonary aneurysms with anti–tumor necrosis factor treatment
Author(s) -
Ghirardo Sergio,
Pastore Serena,
Gortani Giulia,
Tommasini Alberto,
Taddio Andrea
Publication year - 2019
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/ppul.24298
Subject(s) - medicine , infliximab , tumor necrosis factor α , tumor necrosis factor alpha , cyclophosphamide , regimen , surgery , chemotherapy
We report the first case of Hughes Stovin Syndrome successfully treated with long‐lasting tumor necrosis factor α (TNF‐α) blocker (infliximab) treatment. Because of the failure of the standard therapeutic regimen with steroids and cyclophosphamide, infliximab was started achieving a stable disease remission and a complete resolution of pulmonary aneurysms. Hughes Stovin Syndrome, although rare, is a life‐threatening condition that needs to be timely identified and treated aggressively. Our report underlines the importance of TNF‐α blocker treatment in Hughes Stovin Syndrome, suggesting its use as long‐term safe and useful.