Clinical features and management of children with primary ciliary dyskinesia in England.


Journal

Archives of disease in childhood
ISSN: 1468-2044
Titre abrégé: Arch Dis Child
Pays: England
ID NLM: 0372434

Informations de publication

Date de publication:
08 2020
Historique:
received: 14 06 2019
revised: 26 11 2019
accepted: 03 02 2020
pubmed: 12 3 2020
medline: 21 10 2020
entrez: 12 3 2020
Statut: ppublish

Résumé

In England, the National Health Service commissioned a National Management Service for children with primary ciliary dyskinesia (PCD). The aims of this study were to describe the health of children seen in this Service and compare lung function to children with cystic fibrosis (CF). Multi-centre service evaluation of the English National Management PCD Service. Four nationally commissioned PCD centres in England. 333 children with PCD reviewed in the Service in 2015; lung function data were also compared with 2970 children with CF. Median age at diagnosis for PCD was 2.6 years, significantly lower in children with situs inversus (1.0 vs 6.0 years, p<0.001). Compared with national data from the CF Registry, mean (SD) %predicted forced expiratory volume in one second (FEV We provide evidence that children with PCD in England have worse lung function than those with CF. Nutritional status should be considered in PCD management, as those with a lower BMI have significantly lower FEV

Identifiants

pubmed: 32156696
pii: archdischild-2019-317687
doi: 10.1136/archdischild-2019-317687
doi:

Types de publication

Comparative Study Journal Article Multicenter Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

724-729

Investigateurs

Amanda Harris (A)
Amanda Friend (A)
Victoria Keenan (V)
Hannah Wilkins (H)
Hasnaa Ismail-Koch (H)
Andrea Burgess (A)
Samantha Packham (S)
Patricia Goggin (P)
Gemma Marsh (G)
Laura Baynton (L)
Michael Leshen (M)
Sarah Ollosson (S)
Nicola Collins (N)
Peter Rea (P)
Ann-Louise McDermott (AL)
Amanda Parsons (A)
Rajinder Kang (R)
Nicola Lloyd (N)
Alison Truscott (A)
Lynne Schofield (L)
L Knight (L)

Informations de copyright

© Author(s) (or their employer(s)) 2020. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: JL reports grants, personal fees and non-financial support from Aerocrine/ Circassia, grants and personal fees from Vertex, grants from Parion, outside the submitted work. SC reports grants, personal fees and non-financial support from Vertex Pharmaceuticals, grants and other from Chiesi Pharmaceuticals, other from Pharmaxis Pharmaceuticals, personal fees from Actavis Pharmaceuticals, other from Profile Pharma, outside the submitted work.

Auteurs

Bruna Rubbo (B)

School of Clinical and Experimental Medicine, Faculty of Medicine, University of Southampton, Southampton, UK.
Primary Ciliary Dyskinesia Centre, NIHR Southampton Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK.

Sunayna Best (S)

Department of Paediatric Respiratory Medicine, Paediatric Primary Ciliary Dyskinesia Centre, Royal Brompton and Harefield NHS Foundation Trust, London, UK.

Robert Anthony Hirst (RA)

PCD Diagnosis and Research, Department of Respiratory Sciences, University of Leicester, Leicester, UK.

Amelia Shoemark (A)

Department of Paediatric Respiratory Medicine, Paediatric Primary Ciliary Dyskinesia Centre, Royal Brompton and Harefield NHS Foundation Trust, London, UK.

Patricia Goggin (P)

School of Clinical and Experimental Medicine, Faculty of Medicine, University of Southampton, Southampton, UK.
Primary Ciliary Dyskinesia Centre, NIHR Southampton Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK.

Siobhan B Carr (SB)

Department of Paediatric Respiratory Medicine, Paediatric Primary Ciliary Dyskinesia Centre, Royal Brompton and Harefield NHS Foundation Trust, London, UK.
National Heart and Lung Institute, Imperial College, London, UK.

Philip Chetcuti (P)

Respiratory Paediatrics, Leeds Teaching Hospitals NHS Trust, Leeds, UK.

Claire Hogg (C)

Department of Paediatric Respiratory Medicine, Paediatric Primary Ciliary Dyskinesia Centre, Royal Brompton and Harefield NHS Foundation Trust, London, UK.
National Heart and Lung Institute, Imperial College, London, UK.

Priti Kenia (P)

Department of Respiratory Medicine, Birmingham Women's and Children's NHS Foundation Trust, Birmingham, UK.

Jane S Lucas (JS)

Primary Ciliary Dyskinesia Centre, NIHR Southampton Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK.

Eduardo Moya (E)

Paediatric Department, Bradford Teaching Hospitals NHS Foundation Trust, Bradford, UK.

Manjith Narayanan (M)

PCD Diagnosis and Research, Department of Respiratory Sciences, University of Leicester, Leicester, UK.
Leicester National Primary Ciliary Dyskinesia diagnosis and management service, University Hospitals of Leicester NHS Trust, Leicester, UK.

Christopher O'Callaghan (C)

Leicester National Primary Ciliary Dyskinesia diagnosis and management service, University Hospitals of Leicester NHS Trust, Leicester, UK.
UCL Great Ormond Street Institute of Child Health, GOSH NIHR BRC, London, United Kingdom.

Michael Williamson (M)

Leicester National Primary Ciliary Dyskinesia diagnosis and management service, University Hospitals of Leicester NHS Trust, Leicester, UK.

Woolf Theodore Walker (WT)

Primary Ciliary Dyskinesia Centre, NIHR Southampton Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK woolf.walker@uhs.nhs.uk.

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Classifications MeSH