Alexandru, Mihaela; Veil, Raphaël; Rubbo, Bruna; Goutaki, Myrofora; Kim, Sookyung; Lam, Yin Ting; Nevoux, Jérôme; Lucas, Jane S; Papon, Jean-François (2023). Ear and upper airway clinical outcome measures for use in primary ciliary dyskinesia research: a scoping review. European respiratory review, 32(169), p. 220200. European Respiratory Society 10.1183/16000617.0200-2022
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BACKGROUND
Primary ciliary dyskinesia (PCD) is a rare genetic disorder characterised by pulmonary, otological and sino-nasal manifestations. Well-defined clinical outcome measures are needed in such rare diseases research to improve follow-up and treatments. Pulmonary outcome measures have recently been described. The aim of this study was to identify ear and upper airway outcome measures that could be used for longitudinal follow-up of individuals with PCD.
METHODS
A scoping review was performed by systematically searching MEDLINE, Embase and Cochrane Database of Systematic Reviews online databases for studies published from January 1996 to March 2022 that included at least 10 adult or paediatric PCD patients and reported ear and upper airway outcomes.
RESULTS
33 studies (1794 patients) were included. 10 ear and upper airway outcomes were reported. 17 studies reported audiometry, 16 reported otoscopic findings, and 13 reported rhinoscopic findings and sinus imaging. Health-related quality of life questionnaires were performed in seven studies. There was a high variability in definitions and measurement of outcomes between studies.
CONCLUSIONS
This scoping review highlights the lack of data regarding ear and upper airway outcomes in PCD. It also reports a high heterogeneity in outcome definitions or measures. We provide well-founded specific suggestions to standardise ear and upper airway outcome definitions and reporting for future PCD research studies.
Item Type: |
Journal Article (Review Article) |
|---|---|
Division/Institute: |
04 Faculty of Medicine > Department of Gynaecology, Paediatrics and Endocrinology (DFKE) > Clinic of Paediatric Medicine 04 Faculty of Medicine > Pre-clinic Human Medicine > Institute of Social and Preventive Medicine (ISPM) 04 Faculty of Medicine > Department of Gynaecology, Paediatrics and Endocrinology (DFKE) > Clinic of Paediatric Medicine > Paediatric Pneumology |
Graduate School: |
Graduate School for Cellular and Biomedical Sciences (GCB) |
UniBE Contributor: |
Goutaki, Myrofora, Lam, Yin Ting |
Subjects: |
600 Technology > 610 Medicine & health 300 Social sciences, sociology & anthropology > 360 Social problems & social services |
ISSN: |
0905-9180 |
Publisher: |
European Respiratory Society |
Language: |
English |
Submitter: |
Pubmed Import |
Date Deposited: |
13 Jul 2023 10:21 |
Last Modified: |
11 Jan 2024 14:47 |
Publisher DOI: |
10.1183/16000617.0200-2022 |
PubMed ID: |
37437912 |
BORIS DOI: |
10.48350/184731 |
URI: |
https://boris.unibe.ch/id/eprint/184731 |
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