ArticlePloS one2014
Culture of primary ciliary dyskinesia epithelial cells at air-liquid interface can alter ciliary phenotype but remains a robust and informative diagnostic aid.
Article in PloS one, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 50 papers, 2 of them syntheses that pooled it.
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Who cites it
50 citing papers in PubMed, 2 syntheses or guidelines pooled it, 115 citations in OpenAlex.
- Diagnosis of Primary Ciliary Dyskinesia. An Official American Thoracic Society Clinical Practice Guideline.American journal of respiratory and critical care medicine · 2018Pooled it
- European Respiratory Society guidelines for the diagnosis of primary ciliary dyskinesia.The European respiratory journal · 2017Guideline
- Utilising human cellular models of primary ciliary dyskinesia: a scoping review.European respiratory review : an official journal of the European Respiratory Society · 2026Article
- Preclinical human models of primary ciliary dyskinesia.European respiratory review : an official journal of the European Respiratory Society · 2026Review
- CFAP300 Loss-of-Function Mutations with Primary Ciliary Dyskinesia: Evidence from Ex Vivo and ALI Cultures.International journal of molecular sciences · 2025Article
- Cross-modal predictive modeling of multi-omic data in 3D airway organ tissue equivalents during viral infection.Frontiers in genetics · 2025Article
- A novelThe European respiratory journal · 2024Article
- Pathogenic variants inThe European respiratory journal · 2024Article
- Primary Ciliary Dyskinesia: A Clinical Review.Cells · 2024Review
- Advancing Primary Ciliary Dyskinesia Diagnosis through High-Speed Video Microscopy Analysis.Cells · 2024Article
- Exploring the host response in infected lung organoids using NanoString technology: A statistical analysis of gene expression data.PloS one · 2024Article
- Development of a novel air-liquid interface airway tissue equivalent model for in vitro respiratory modeling studies.Scientific reports · 2023Article
- Comparing Cytology Brushes for Optimal Human Nasal Epithelial Cell Collection: Implications for Airway Disease Diagnosis and Research.Journal of personalized medicine · 2023Article
- TheInternational journal of molecular sciences · 2023Review
- Clinical implications of respiratory ciliary dysfunction in heterotaxy patients with congenital heart disease: elevated risk of postoperative airway complications.Frontiers in cardiovascular medicine · 2023Article
- Primary ciliary dyskinesia diagnosis and management and its implications in America: a mini review.Frontiers in pediatrics · 2023Review
- NovelJournal of clinical medicine · 2022Article
- PCD Genes-From Patients to Model Organisms and Back to Humans.International journal of molecular sciences · 2022Review
- Temporal Whole-Transcriptomic Analysis of CharacterizedFrontiers in cell and developmental biology · 2022Article
- Article
Corrections and comments
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Authors and funding
12 authors at 6 institutions in 1 country.
Funding
Abstract
backgroundThe diagnosis of primary ciliary dyskinesia (PCD) requires the analysis of ciliary function and ultrastructure. Diagnosis can be complicated by secondary effects on cilia such as damage during sampling, local inflammation or recent infection. To differentiate primary from secondary abnormalities, re-analysis of cilia following culture and re-differentiation of epithelial cells at an air-liquid interface (ALI) aids the diagnosis of PCD. However changes in ciliary beat pattern of cilia following epithelial cell culture has previously been described, which has brought the robustness of this method into question. This is the first systematic study to evaluate ALI culture as an aid to diagnosis of PCD in the light of these concerns.
methodsWe retrospectively studied changes associated with ALI-culture in 158 subjects referred for diagnostic testing at two PCD centres. Ciliated nasal epithelium (PCD n = 54; non-PCD n 111) was analysed by high-speed digital video microscopy and transmission electron microscopy before and after culture.
resultsCiliary function was abnormal before and after culture in all subjects with PCD; 21 PCD subjects had a combination of static and uncoordinated twitching cilia, which became completely static following culture, a further 9 demonstrated a decreased ciliary beat frequency after culture. In subjects without PCD, secondary ciliary dyskinesia was reduced.
conclusionsThe change to ciliary phenotype in PCD samples following cell culture does not affect the diagnosis, and in certain cases can assist the ability to identify PCD cilia.
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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.