Hypersensitivity pneumonitis: Lessons from a randomized controlled trial in children

Abstract

Introduction: Hypersensitivity pneumonitis (HP) in children is a severe interstitial lung disease and potentially, a chronic condition, if not treated appropriately. No evidence-based guidelines are available; in particular, the role of systemic glucocorticoid therapy is unclear.Methods: The aim of this randomized, double-blind, placebo-controlled, parallelgroup, multi-center, phase II trial in pediatric HP was to assess the outcome of HP in children after 6 months of treatment and to compare 3 months of treatment with oral prednisolone or placebo.Results: After 1.5 years and the inclusion of only four children, we terminated the study prematurely. Two of the children randomized to prednisolone did not achieve the predefined response of FVC to normal. One child treated with placebo recovered to normal, similar to another child treated with prednisolone. All children treated with steroids developed drug-related side effects.Discussion: This uncompleted study illustrates the urgent medical need for evidence-based treatment protocols for this condition. We discuss the hurdles which were specific for completion of this trial in a rare condition. Among other options, we suggest the inclusion of children into an all-age study of HP, as in adults the same questions are unanswered.This is an open access article under the terms of the Creative Commons Attribution-NonCommercial License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited and is not used for commercial purposes.

Pediatric hypersensitivity pneumonitis: literature update and proposal of a diagnostic algorithm

“…An early diagnosis is crucial in children [ 43 , 44 ]. In case of delayed diagnosis and treatment, HP can convert into a chronic and progressive form, and it can also be fatal (29% of cases at 5 years) [ 44 – 47 ].

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“…1 ). Diffuse ground-glass opacity and hyperdensity are the most significant radiological signs [ 41 ]; 96% of the HRCT scans showed characteristic nodular opacities, 5% linear opacities, and 73% ground-glass pattern with increased attenuation [ 44 ]. In the study of Buchvald et al, all 19 patients had abnormal HRCT at the time of diagnosis with a diffusely increased attenuation of the parenchyma [ 7 ].…”

“…However, the lack of randomized controlled trials in children makes the role of systemic glucocorticoid therapy unclear. In addition, no evidence-based guidelines are available [ 44 ]. Consequently, childhood interstitial lung disease (chILD) guidelines represent the reference, especially in children ventilated or closed to ventilation.…”

Acute respiratory distress after metofluthrin insecticide ingestion in a 19-month-old girl

“…또한, 증상이 지속하거나 폐기능 또는 고해 상컴퓨터단층촬영에서 심각한 손상이 있다면, methylprednisolone 충격요법(pulse therapy)과 같이 스테로 이드 정맥투여를 고려할 수 있다 15) . 그러나, 아직 소아 스 테로이드 요법에 관한 지침 및 근거가 명확하지 않은 상태 이다 15,19) .…”