Neil J Hime1,2, Yvonne Zurynski1,2, Dominic Fitzgerald3,4, Hiran Selvadurai3,4, Amy Phu1,2, Marie Deverell1,2, Elizabeth J Elliott1,2,5, Adam Jaffe6,7. 1. Discipline of Paediatrics and Child Health, Sydney Medical School, The University of Sydney, Sydney, Australia. 2. Australian Paediatric Surveillance Unit, Kids Research Institute, Westmead NSW 2145, Australia. 3. Department of Respiratory Medicine, The Children's Hospital at Westmead, Westmead NSW 2145, Australia. 4. Discipline of Paediatrics and Child Health, Sydney Medical School, The University of Sydney Clinical School, Sydney, Australia. 5. The Children's Hospital at Westmead, Sydney, Australia. 6. Department of Respiratory Medicine, Sydney Children's Hospital, Randwick NSW 2031, Australia. 7. Discipline of Paediatrics, School of Women's and Children's Health, UNSW Medicine, The University of New South Wales, Sydney, Australia.
Abstract
OBJECTIVES: Childhood interstitial lung disease (chILD) is a group of rare chronic and complex disorders of variable pathology. There has been no systematic review of published chILD research. This study aimed to describe chILD classification systems, epidemiology, morbidity, treatments, outcomes, and the impact of chILD on families and the burden on health services. METHODS: A systematic literature search for original studies on chILD was undertaken in the major biomedical databases to the end of December 2013. Epidemiological studies, case series and studies describing classification systems were included. Single case studies were excluded. RESULTS: The search yielded 37 publications that met study criteria. Four different chILD classification systems have been proposed in the past decade. The incidence of chILD has been estimated at 0.13-16.2 cases/100,000 children/year. One to five new cases presented to individual hospitals each year. In developed countries, the median mortality was 13% (6-19%). Morbidity and outcomes were highly variable and not systematically reported. Corticosteroids and hydroxychloroquine were the most common treatments. The impact of chILD on families and the burden on health services has not been studied. CONCLUSIONS: The heterogeneity of the chILD group of disorders, different determinations of what constitutes a chILD disorder and, a paucity of large epidemiological studies precludes consolidation of results across studies. Consensus on chILD classification is needed to support diagnosis and allow direct comparisons of research evidence. Active disease surveillance and international patient registries are required to advance understanding and management of chILD.
OBJECTIVES: Childhood interstitial lung disease (chILD) is a group of rare chronic and complex disorders of variable pathology. There has been no systematic review of published chILD research. This study aimed to describe chILD classification systems, epidemiology, morbidity, treatments, outcomes, and the impact of chILD on families and the burden on health services. METHODS: A systematic literature search for original studies on chILD was undertaken in the major biomedical databases to the end of December 2013. Epidemiological studies, case series and studies describing classification systems were included. Single case studies were excluded. RESULTS: The search yielded 37 publications that met study criteria. Four different chILD classification systems have been proposed in the past decade. The incidence of chILD has been estimated at 0.13-16.2 cases/100,000 children/year. One to five new cases presented to individual hospitals each year. In developed countries, the median mortality was 13% (6-19%). Morbidity and outcomes were highly variable and not systematically reported. Corticosteroids and hydroxychloroquine were the most common treatments. The impact of chILD on families and the burden on health services has not been studied. CONCLUSIONS: The heterogeneity of the chILD group of disorders, different determinations of what constitutes a chILD disorder and, a paucity of large epidemiological studies precludes consolidation of results across studies. Consensus on chILD classification is needed to support diagnosis and allow direct comparisons of research evidence. Active disease surveillance and international patient registries are required to advance understanding and management of chILD.
Authors: Paul E Moore; Jason T Poston; Debra Boyer; Emily Barsky; Jonathan Gaffin; Kathleen B Boyne; Kristie R Ross; Laura Beth Mann Dosier; Timothy J Vece; Alicia M Casey; Sebastian K Welsh; J Wells Logan; Edward G Shepherd; Pelton A Phinzy; Howard B Panitch; Christina M Papantonakis; Eric D Austin; Amir B Orandi; Maleewan Kitcharoensakkul; Mark K Abe; Amjad Horani; Jordan S Rettig; Jessica Pittman Journal: Ann Am Thorac Soc Date: 2017-08
Authors: Robin R Deterding; Emily M DeBoer; Michal J Cidon; Terry E Robinson; David Warburton; Gail H Deutsch; Lisa R Young Journal: Am J Respir Crit Care Med Date: 2019-11-15 Impact factor: 21.405
Authors: Matthew M Willmering; Laura L Walkup; Peter J Niedbalski; Hui Wang; Ziyi Wang; Erik B Hysinger; Kasiani C Myers; Christopher T Towe; Bastiaan Driehuys; Zackary I Cleveland; Jason C Woods Journal: J Magn Reson Imaging Date: 2022-03-04 Impact factor: 5.119