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Time trends in diagnostic testing for primary ciliary dyskinesia in Europe

Florian S. Halbeisen, Amelia Shoemark, Angelo Barbato, Mieke Boon, Siobhan Carr, Suzanne Crowley, Rob Hirst, Bulent Karadag, Cordula Koerner-Rettberg, Michael R. Loebinger, Jane S. Lucas, Bernard Maitre, Henryk Mazurek, Uğur Özçelik, Vendula Martinů, Nicolaus Schwerk, Guillaume Thouvenin, Stefan A. Tschanz, Panayiotis Yiallouros, Myrofora Goutaki, Claudia E. Kuehni
European Respiratory Journal 2019 54: 1900528; DOI: 10.1183/13993003.00528-2019
Florian S. Halbeisen
Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland
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  • ORCID record for Florian S. Halbeisen
Amelia Shoemark
Dept of Paediatrics, Primary Ciliary Dyskinesia Centre, Royal Brompton and Harefield Foundation Trust, London, UKSchool of Medicine, University of Dundee, Dundee, UK
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Angelo Barbato
Italian PCD ConsortiumDept of Paediatrics, University of Padova, Padova, Italy
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Mieke Boon
Dept of Paediatrics, University Hospital Gasthuisberg, Leuven, Belgium
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Siobhan Carr
Dept of Paediatrics, Primary Ciliary Dyskinesia Centre, Royal Brompton and Harefield Foundation Trust, London, UK
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  • ORCID record for Siobhan Carr
Suzanne Crowley
Unit for Paediatric Heart, Lung, Allergic Diseases, Rikshospitalet, Oslo, Norway
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Rob Hirst
Dept of Infection, Immunity and Inflammation, Institute for Lung Health, University of Leicester, Leicester, UK
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Bulent Karadag
Dept of Pediatric Pulmonology, Marmara University, School of Medicine, Istanbul, Turkey
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Cordula Koerner-Rettberg
Dept of Paediatric Pneumology, University Children's Hospital of Ruhr University Bochum, Bochum, Germany
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Michael R. Loebinger
Host Defence Unit, Royal Brompton and Harefield NHS Foundation Trust, London, UK
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Jane S. Lucas
Primary Ciliary Dyskinesia Centre, NIHR Respiratory Biomedical Research Centre, University of Southampton, Southampton, UK
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  • ORCID record for Jane S. Lucas
Bernard Maitre
French Reference Centre for Rare Lung DiseasesHopital intercommunal de Créteil, Service de Pneumologie, DHU ATVB, Université Paris Est Créteil, Paris, France
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Henryk Mazurek
Dept of Pneumonology and Cystic Fibrosis, Institute of Tuberculosis and Lung Disorders, Zdrój, Poland
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Uğur Özçelik
Dept of Pediatric Pulmonology, Hacettepe University Faculty of Medicine, Ankara, Turkey
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Vendula Martinů
Pediatric Dept, Charles University Prague and University Hospital Motol, Prague, Czech Republic
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Nicolaus Schwerk
Clinic for Paediatric Pulmonology, Allergiology and Neonatology, Hannover Medical School, Hannover, Germany
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Guillaume Thouvenin
French Reference Centre for Rare Lung DiseasesPaediatric Pulmonary Dept, Trousseau Hospital APHP, Sorbonne Universities and Pierre et Marie Curie University, Paris, FranceSorbonne Université, INSERM, Centre de Recherche Saint-Antoine, CRSA, Paris, France
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Stefan A. Tschanz
Swiss PCD registry (CH-PCD)Institute of Anatomy, University of Bern, Bern, Switzerland
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Panayiotis Yiallouros
Medical School, University of Cyprus, Nicosia, Cyprus
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Myrofora Goutaki
Institute of Social and Preventive Medicine, University of Bern, Bern, SwitzerlandPaediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Bern, SwitzerlandBoth authors contributed equally
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Claudia E. Kuehni
Institute of Social and Preventive Medicine, University of Bern, Bern, SwitzerlandPaediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Bern, SwitzerlandBoth authors contributed equally
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  • For correspondence: claudia.kuehni@ispm.unibe.ch
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Extract

Despite recent advances in diagnostic methods, diagnosis of primary ciliary dyskinesia (PCD) remains complex. We need a combination of different diagnostic tests, and all have their limitations [1]. In 2009, the first European Respiratory Society (ERS) Task Force on PCD in children published recommendations [2], suggesting that: 1) nasal nitric oxide (nNO) should be measured to screen for PCD in patients aged ≥5 years [3]; and 2) video microscopy (VM) analysis of ciliary beat pattern and frequency [4] plus electron microscopy (EM) [5] should be the key confirmatory diagnostic tests. Genetic testing was not recommended as part of the initial diagnostic testing, but as an additional test for inconclusive cases. The recommended test combination was nNO, VM and EM for patients aged ≥5 years and VM plus EM for younger patients.

Abstract

Adherence to the 2009 ERS task force diagnostic recommendations was low. To further improve PCD diagnosis, we must be more diligent and engaging in implementing the new evidence-based guidelines published in 2017. http://bit.ly/2zvjpBh

Footnotes

  • Conflict of interest: F.S. Halbeisen has nothing to disclose.

  • Conflict of interest: A. Shoemark has nothing to disclose.

  • Conflict of interest: A. Barbato has nothing to disclose.

  • Conflict of interest: M. Boon reports grants from Horizon 2020, outside the submitted work.

  • Conflict of interest: S. Carr reports personal fees for advisory board work, acting as registry trial PI, and travel and accommodation expenses for meetings from Vertex Pharmaceuticals, advisory board honoraria from Chiesi Pharmaceuticals, personal fees for lectures from Actavis Pharmaceuticals, and has acted as trial PI for Pharmaixis Pharmaceuticals, outside the submitted work.

  • Conflict of interest: S. Crowley has nothing to disclose.

  • Conflict of interest: R. Hirst has nothing to disclose.

  • Conflict of interest: B. Karadag has nothing to disclose.

  • Conflict of interest: C. Koerner-Rettberg has nothing to disclose.

  • Conflict of interest: M.R. Loebinger reports personal fees for advisory board work from Bayer, Polyphor and Raptor, personal fees for advisory board work lectures from Griffols, and is UK chief investigator for Parion, outside the submitted work.

  • Conflict of interest: J.S. Lucas reports grants, personal fees for advisory board work, non-financial support for meeting attendance and equipment donations from Aerocrine/Circassia, grants and personal fees for advisory board work from Vertex, grants from Parion, outside the submitted work.

  • Conflict of interest: B. Maitre has nothing to disclose.

  • Conflict of interest: H. Mazurek has nothing to disclose.

  • Conflict of interest: U. Ozcelik has nothing to disclose.

  • Conflict of interest: V. Martinu has nothing to disclose.

  • Conflict of interest: N. Schwerk has nothing to disclose.

  • Conflict of interest: G. Thouvenin has nothing to disclose.

  • Conflict of interest: S.A. Tschanz has nothing to disclose

  • Conflict of interest: P. Yiallouros has nothing to disclose.

  • Conflict of interest: M. Goutaki has nothing to disclose.

  • Conflict of interest: C.E. Kuehni has nothing to disclose.

  • Support statement: This work was funded by Schweizerischer Nationalfonds zur Förderung der Wissenschaftlichen Forschung (grant 320030_173044). Funding information for this article has been deposited with the Crossref Funder Registry.

  • Received March 14, 2019.
  • Accepted June 11, 2019.
  • Copyright ©ERS 2019
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Time trends in diagnostic testing for primary ciliary dyskinesia in Europe
Florian S. Halbeisen, Amelia Shoemark, Angelo Barbato, Mieke Boon, Siobhan Carr, Suzanne Crowley, Rob Hirst, Bulent Karadag, Cordula Koerner-Rettberg, Michael R. Loebinger, Jane S. Lucas, Bernard Maitre, Henryk Mazurek, Uğur Özçelik, Vendula Martinů, Nicolaus Schwerk, Guillaume Thouvenin, Stefan A. Tschanz, Panayiotis Yiallouros, Myrofora Goutaki, Claudia E. Kuehni
European Respiratory Journal Oct 2019, 54 (4) 1900528; DOI: 10.1183/13993003.00528-2019

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Time trends in diagnostic testing for primary ciliary dyskinesia in Europe
Florian S. Halbeisen, Amelia Shoemark, Angelo Barbato, Mieke Boon, Siobhan Carr, Suzanne Crowley, Rob Hirst, Bulent Karadag, Cordula Koerner-Rettberg, Michael R. Loebinger, Jane S. Lucas, Bernard Maitre, Henryk Mazurek, Uğur Özçelik, Vendula Martinů, Nicolaus Schwerk, Guillaume Thouvenin, Stefan A. Tschanz, Panayiotis Yiallouros, Myrofora Goutaki, Claudia E. Kuehni
European Respiratory Journal Oct 2019, 54 (4) 1900528; DOI: 10.1183/13993003.00528-2019
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