Evidence map›Paper›PMID 37437912›Full record

ArticleEuropean respiratory review : an official journal of the European Respiratory Society2023

Ear and upper airway clinical outcome measures for use in primary ciliary dyskinesia research: a scoping review.

Mihaela Alexandru, Raphaël Veil, Bruna Rubbo, Myrofora Goutaki, Sookyung Kim, Yin Ting Lam, Jérôme Nevoux, Jane S Lucas, Jean-François Papon

Abstract readScoping Review
In one paragraph

Article in European respiratory review : an official journal of the European Respiratory Society, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Article
  2. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors.

Mihaela AlexandruAP-HP, Université Paris-Saclay, Hôpital Bicêtre, Service d'ORL, Le Kremlin-Bicêtre, France mihaela.dana.alexandru@gmail.com.ORCID https://orcid.org/0000-0001-5134-7344
Raphaël VeilAP-HP, Université Paris-Saclay, Hôpital Bicêtre, Service d'Épidémiologie et Santé Publique, Le Kremlin-Bicêtre, France.
Bruna RubboPrimary Ciliary Dyskinesia Centre, NIHR Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK.
Myrofora GoutakiInstitute of Social and Preventive Medicine, University of Bern, Bern, Switzerland.ORCID https://orcid.org/0000-0001-8036-2092
Sookyung KimAP-HP, Université Paris-Saclay, Hôpital Bicêtre, Service d'ORL, Le Kremlin-Bicêtre, France.
Yin Ting LamInstitute of Social and Preventive Medicine, University of Bern, Bern, Switzerland.ORCID https://orcid.org/0000-0002-2380-834X
Jérôme NevouxAP-HP, Université Paris-Saclay, Hôpital Bicêtre, Service d'ORL, Le Kremlin-Bicêtre, France.
Jane S LucasPrimary Ciliary Dyskinesia Centre, NIHR Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK.ORCID https://orcid.org/0000-0001-8701-9975
Jean-François PaponAP-HP, Université Paris-Saclay, Hôpital Bicêtre, Service d'ORL, Le Kremlin-Bicêtre, France.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundPrimary 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.

methodsA 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.

results33 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.

conclusionsThis 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.

Indexed as

Ciliary Motility DisordersQuality of LifeAdultChildHumansRare Diseases

Identifiers

PMID37437912
PMCPMC10336562

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.