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Record W4407846353 · doi:10.1002/oto2.70079

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

2025· article· en· W4407846353 on OpenAlexaff
Isabelle A. Dagher, Adam J. Kimple, Thomas W. Ferkol, Scott D. Sagel, Sharon Dell, Carlos Milla, Lang Li, Feng‐Chang Lin, Kelli M. Sullivan, Maimoona A. Zariwala, Michael R. Knowles, Margaret Rosenfeld, Margaret W. Leigh, Stephanie D. Davis

Bibliographic record

VenueOTO Open · 2025
Typearticle
Languageen
FieldMedicine
TopicCystic Fibrosis Research Advances
Canadian institutionsBC Children's HospitalSickKids Foundation
FundersNational Heart, Lung, and Blood Institute
KeywordsPrimary ciliary dyskinesiaMedicineSinusitisHearing lossPediatricsOtorhinolaryngologyNoseChronic sinusitisNasal congestionCohortLongitudinal studyMiddle earCohort studySurgeryAudiologyPathologyInternal medicineBronchiectasisLung

Abstract

fetched live from OpenAlex

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.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.113
Threshold uncertainty score0.317

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.001
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.011
GPT teacher head0.364
Teacher spread0.353 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations5
Published2025
Admission routes1
Has abstractyes

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