Article Text

Download PDFPDF
Original research
Healthcare resource utilisation and medical costs for children with interstitial lung diseases (chILD) in Europe
  1. Elias Seidl1,
  2. Nicolaus Schwerk2,
  3. Julia Carlens2,
  4. Martin Wetzke2,
  5. Steve Cunningham3,
  6. Nagehan Emiralioğlu4,
  7. Nural Kiper4,
  8. Joanna Lange5,
  9. Katarzyna Krenke5,
  10. Nicola Ullmann6,
  11. Dora Krikovszky7,
  12. the chILD-EU collaborators,
  13. Phillen Maqhuzu8,9,
  14. Charlotte A Griese10,
  15. Larissa Schwarzkopf8,9,11,
  16. Matthias Griese1,9
  1. 1Department of Pediatrics, Dr. von Hauner Children's Hospital, University Hospital, LMU, Munich, Germany
  2. 2Department of Pediatric Pneumology, Allergology, and Neonatology, Hannover Medical School, German Center for Lung Research, Hannover, Germany
  3. 3Department of Child Life and Health, Centre for Inflammation Research, University of Edinburgh, Edinburgh, UK
  4. 4Department of Pediatric Pulmonology, School of Medicine, Hacettepe University Faculty of Medicine, Ankara, Turkey
  5. 5Department of Pediatric Pneumology and Allergy, Warszawski Uniwersytet Medyczny, Warszawa, Poland
  6. 6Pediatric Pulmonology and Respiratory Intermediate Care Unit, Sleep and Long Term Ventilation Unit, Academic Department of Pediatrics (DPUO), Pediatric Hospital “Bambino Gesù” Research Institute, Rome, Italy
  7. 7First Department of Paediatrics, Semmelweis University, Budapest, Hungary
  8. 8Institute of Health Economics and Health Care Management, Helmholtz Zentrum München, German Research Center for Environmental Health, Neuherberg, Germany
  9. 9Comprehensive Pneumology Center-Munich (CPC-M), Member of the German Center for Lung Diseases DZL, Munich, Germany
  10. 10Georg-August-Universität Göttingen, Faculty of Business and Economics, Göttingen, Germany
  11. 11Institute of Health Economics and Health Care Management, Helmholtz Zentrum Munchen Deutsches Forschungszentrum fur Umwelt und Gesundheit, Neuherberg, Germany
  1. Correspondence to Dr Matthias Griese, Paediatrics, Munich University Hospital Dr von Hauner Children's Hospital, Munchen, Bayern, Germany; matthias.griese{at}med.uni-muenchen.de

Abstract

Background No data on healthcare utilisation and associated costs for the many rare entities of children’s interstitial lung diseases (chILD) exist. This paper portrays healthcare utilisation structures among individuals with chILD, provides a pan-European estimate of a 3-month interval per-capita costs and delineates crucial cost drivers.

Methods Based on longitudinal healthcare resource utilisation pattern of 445 children included in the Kids Lung Register diagnosed with chILD across 10 European countries, we delineated direct medical and non-medical costs of care per 3-month interval. Country-specific utilisation patterns were assessed with a children-tailored modification of the validated FIMA questionnaire and valued by German unit costs. Costs of care and their drivers were subsequently identified via gamma-distributed generalised linear regression models.

Results During the 3 months prior to inclusion into the registry (baseline), the rate of hospital admissions and inpatient days was high. Unadjusted direct medical per capita costs (€19 818) exceeded indirect (€1 907) and direct non-medical costs (€1 125) by far. Country-specific total costs ranged from €8 713 in Italy to €28 788 in Poland. Highest expenses were caused by the disease categories ‘diffuse parenchymal lung disease (DPLD)-diffuse developmental disorders’ (€45 536) and ‘DPLD-unclear in the non-neonate’ (€47 011). During a follow-up time of up to 5 years, direct medical costs dropped, whereas indirect costs and non-medical costs remained stable.

Conclusions This is the first prospective, longitudinal study analysing healthcare resource utilisation and costs for chILD across different European countries. Our results indicate that chILD is associated with high utilisation of healthcare services, placing a substantial economic burden on health systems.

  • paediatric interstitial lung disease
  • paediatric lung disaese

Data availability statement

Data are available upon reasonable request.

Statistics from Altmetric.com

Request Permissions

If you wish to reuse any or all of this article please use the link below which will take you to the Copyright Clearance Center’s RightsLink service. You will be able to get a quick price and instant permission to reuse the content in many different ways.

Data availability statement

Data are available upon reasonable request.

View Full Text

Footnotes

  • LS and MG are joint senior authors.

  • LS and MG contributed equally.

  • Collaborators The chILD-EU collaborators are Tugba Sysmanlar, Ayse Aslan (Ankara Gazi University, Turkey), Susanne Zeidler (Asklepios Sankt Augustin Krankenhaus, Germany), Veronika Baumeister, Folke Brinkmann (Bochum Ruhr Universität, Germany), Isabelle Rochat (Centre hospitalier universitaire vaudois Lausanne, Switzerland), Sebastian Becker (Darmstädter Kinderkliniken, Germany), Claudia Eismann, Meike Köhler, Dörthe Neuner, Andrea Schams, Waltraud Wesselak, Sabine Witt, Matthias Kappler, Mandeep Kaur (Dr. von Hauner Children’s Hospital, University Hospital LMU Munich, Germany), Silvia Castillo Corullón (Fundación de Investigación del Hospital Clínico Universitario de Valencia, Spain), Lale Bayir, Nagehan Ciftci (Hannover Medical School, Germany), Hildegard Seidl (Helmholzzentrum Munich, Germany), Lauren McCann (Imperial College London, United Kingdom), Chiara Carolina Abbas, Tobias Ehrenreich, Philipp Latzin (Inselspital Bern, Switzerland), Ayse Aizit Kilinc (Istanbul Cerrahpasa University, Medicine Faculty Hospital, Turkey), Tobias Hübner, Holger Köster (Kinderklinik Oldenburg, Germany), Martin Schebek (Klinikum Kassel, Germany), Matteo Fontana, Nicolas Regamey (Luzerner Kantonsspital, Switzerland), Stanislaw Boguslawski, Honorata Marczak (Medical University of Warsaw, Poland), Deborah Adelsberger (Oldenburg Zentrum für Kinder- und Jugendmedizin, Germany), Achim Freihorst (Ostalab Klinikum Aalen, Germany), Frederik Buchvald (Rigshospitalet Copenhagen, Denmark), Gemma Donohoe (Royal Children’s Hospital Manchester, United Kingdom), Morag MacLean, Fiona Redmond (Royal Hospital for Sick Children Edinburgh, United Kingdom), Cordula Koerner-Rettberg (Ruhr University-Bochum, Germany), Abigel Kolonics-Farkas (Semmelweis University, Hungary), Sarah Shortland (Sheffield Children’s Hospital, United Kingdom), Amy Collier (Southampton General Hospital, United Kingdom), Kathrin Seidemann (Städtische Klinikum Karlsruhe, Germany), Lutz Nährlich (Universität Giessen, Germany), Peter Meißner (Universität Ulm, Germany), Susanne Hämmerling, Benjamin Hanebeck (Universitätsklinikum Heidelberg, Germany), Dominik Schöndorf, Olaf Sommerburg (Universitätsklinikum Heidelberg, Germany), Winfried Baden (Universitätsklinikum Tübingen, Germany), Sune Rubak (University Aarhus, Denmark), Steve Turner (University Aberdeen, United Kingdom), Martin Rosewich, Johannes Schulze, Pera-Silvija Jerkic (University Frankfurt/Main, Germany), Christiane Lex (University Göttignen, Germany), Frans De Baets (University Hospital Gent, Belgium), Zsolt Szepfalusi (University Hospital Vienna, Austria), Edyta Glowacka (University Krakau, Poland), Freerk Prenzel (University Leipzig, Germany), Marijke Proesmans (University Leuven, Belgium), Nazan Cobanoglu (University of Ankara, Turkey), Florian Stehling (University of Essen, Germany), Gisela Anthony, Hans Rock (University of Marburg, Germany), Angelo Barbato, Deborah Snijders (University of Padua, Italy), Andrea Heinzmann (Universtätsklinikum Freiburg, Germany).

  • Contributors ES: processed the experimental data, performed the analytic calculations, designed the figures and took the lead in writing the manuscript. ES, MG, NS, JC, MW, SC, NE, NK, JL, KK, NU, DK handed out the questionnaires and entered the data into the Kids Lung Register. ES and MG conceived, implemented and planned the study. PM and LS helped in developing the methodology, data curation, formal analysis and verified the analytical methods. MG supervised the study and acquired the funding. NS, JC, MW, SC, NE, NK, JL, KK, NU, DK, MP, CG, LS and MG contributed to the final manuscript providing critical feedback. MG is the guarantor, accepts full responsibility for the work and/or the conduct of the study, had access to the data, and controlled the decision to publish.

  • Funding This work was supported by the European Register and Biobank on Childhood Interstitial Lung Diseases (chILD-EU register) funded by the European Commission under FP7-HEALTH-2012-INNOVATION-1, HEALTH.2012.2.4.4-2: Observational trials in rare diseases. MG is supported by DFG (Gr 970/9-1), FP7 Coordination of Non‐Community Research Programmes (Gr FP7‐305653‐chILD‐EU), Cost CA (16125 ENTeR-chILD) and European Respiratory Society Clinical Research Collaboration (CRC).

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Author note Participants contributing less than 2% of the analysed health resource utilisation and medical costs are listed under the chILD-EU collaborators.

  • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.

Linked Articles

  • Paediatric lung disease
    Elias Seidl Nicolaus Schwerk Julia Carlens Martin Wetzke Nagehan Emiralioğlu Nural Kiper Joanna Lange Katarzyna Krenke Zsolt Szepfalusi Florian Stehling Winfried Baden Susanne Hämmerling Silvija-Pera Jerkic Marijke Proesmans Nicola Ullmann Frederik Buchvald Katrin Knoflach Matthias Kappler the chILD EU collaborators Matthias Griese
  • Editorial
    Alicia Casey Robin Deterding