Evidence map›Paper›PMID 39989621›Full record

ArticleOTO open

Progression of Otologic and Nasal Symptoms in Primary Ciliary Dyskinesia Throughout Childhood.

Isabelle Dagher, Adam J Kimple, Thomas W Ferkol, Scott D Sagel, Sharon D Dell, Carlos E Milla, Lang Li, Feng-Chang Lin, Kelli M Sullivan, Maimoona A Zariwala and 5 more

Abstract read
In one paragraph

Article in OTO open. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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

1 citing paper in PubMed.

  1. Article
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

15 authors.

Isabelle DagherDepartment of Pediatrics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.ORCID 0000-0002-8338-4585
Adam J KimpleDepartment of Otolaryngology-Head and Neck Surgery University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.ORCID 0000-0003-1670-8401
Thomas W FerkolDepartment of Pediatrics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.ORCID 0000-0003-2477-9357
Scott D SagelDepartment of Pediatrics Children's Hospital Colorado, University of Colorado School of Medicine Aurora Colorado USA.ORCID 0000-0001-6172-4465
Sharon D DellDepartment of Pediatrics BC Children's Hospital Vancouver British Columbia Canada.ORCID 0000-0003-2169-9407
Carlos E MillaDepartment of Pediatrics Stanford University Palo Alto California USA.ORCID 0000-0001-5515-3053
Lang LiDepartment of Biostatistics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.
Feng-Chang LinDepartment of Biostatistics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.ORCID 0000-0002-2638-1775
Kelli M SullivanDepartment of Pediatrics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.ORCID 0000-0001-8564-4726
Maimoona A ZariwalaDepartment of Pediatrics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.ORCID 0000-0003-1619-1393
Michael R KnowlesMarsico Lung Institute University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.
Margaret RosenfeldDepartment of Pediatrics Seattle Children's Hospital, University of Washington School of Medicine Seattle Washington USA.ORCID 0000-0002-8105-614X
Margaret W LeighDepartment of Pediatrics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.ORCID 0000-0002-6420-2459
Stephanie D DavisDepartment of Pediatrics University of North Carolina at Chapel Hill Chapel Hill North Carolina USA.
Genetic Disorders of Mucociliary Clearance Consortium

Funding

Longitudinal Characterization of Respiratory Tract Exacerbations and Treatment Responses in Primary Ciliary DyskinesiaU54HL096458 · NHLBI · UNIV OF NORTH CAROLINA CHAPEL HILL · PI DAVIS, STEPHANIE DUGGINS, FERKOL, THOMAS W · 2009 to 2023
$20.0M
NHLBI NIH HHS U54 HL096458
6 · The paper itself

Abstract

Objective: Primary ciliary dyskinesia (PCD) is characterized by upper and lower airway disease. Multiple studies have demonstrated the progression of pulmonary disease; however, longitudinal changes in the otologic and nasal symptoms have not been well described in patients. This study defines age-related prevalence, age of onset, and age-related trends in self-reported otologic and sinonasal comorbidities in individuals with PCD. Study Design: A prospective, longitudinal, multicenter, observational study spanning up to 12 years. Setting: Six PCD centers in North America. Methods: Inclusion criteria were <19 years of age and a confirmed diagnosis of PCD based on electron microscopy and/or genetics. A standardized medical history questionnaire and physical exam were completed during each study visit. Descriptive statistics were performed for the entire cohort as well as for subgroups based on ciliary ultrastructure. Results: A total of 147 participants were followed for an average of 7.6 ± 3.2 years. Pressure equalization tubes (PETs) were placed in 80%, transient hearing loss was reported in 68%, and persistent hearing loss was reported in 30%. Hearing aids and speech therapy were utilized by 8% and 27%, respectively. PETs were placed earlier in those with inner dynein arm/microtubular disorganization defects than those with outer dynein arm defects. Participants reported chronic nasal congestion in 97%, sinusitis in 87%, and 35% underwent >1 sinus surgery. Conclusion: There is a high prevalence of reported otologic and sinonasal morbidity among people with PCD that begins during early childhood and persists. Further analysis is indicated to evaluate differences over time among participants with varying ultrastructural defects. Level of Evidence: Level 2.

Indexed as

hearing lossimmotile cilia syndromeKartagener's syndromesinusitis

Identifiers

PMID39989621
PMCPMC11843728

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
Read underepoch 390

Registered trials

None linked

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.