Ciliary Beat Frequency and Pattern: An Accessible Tool for the Screening of Primary Ciliary Dyskinesia

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Authors: Élise Kaspi, Julie Mazenq, Adrien Pagin, Rana Mitri-Frangieh, Mohamed Boucékine, Karine Baumstarck, Thomas Radulesco, Justin Michel, Nadine Desmazes Dufeu, J.‐C. Dubus, Patrice Roll, Diane Frankel
Cited by: 0
References: 22

Abstract

Background/Objectives: Primary ciliary dyskinesia (PCD) is a rare inherited disorder caused by dysfunction of motile cilia, leading to chronic respiratory disease. Diagnosis is challenging due to heterogeneous and non-specific clinical manifestations and the absence of a single definitive diagnostic test. Current diagnostic strategies rely on a combination of functional, ultrastructural, and genetic analyses. The objective of this study was to evaluate whether ciliary beat frequency (CBF), combined with ciliary beat pattern (CBP) assessment using digital high-speed video microscopy (DHSV), could serve as an effective first-line screening tool to identify patients requiring further diagnostic investigations. Methods: This single-center retrospective study included 65 patients (52 children and 13 adults) with clinical suspicion of PCD. Ciliary beat analysis was performed on nasal or bronchial samples using DHSV and Sisson–Ammons Video Analysis software. CBF and CBP were assessed and compared between patients with confirmed PCD and those in whom PCD was excluded based on transmission electron microscopy (TEM) and/or molecular genetic analysis. Results: Fifteen patients were diagnosed with PCD. Mean CBF was significantly lower in the PCD group compared with the non-PCD group (3.3 Hz vs. 8.1 Hz; p < 0.001). A CBF cut-off value of 5.25 Hz yielded a sensitivity of 78.6% and a specificity of 95.7%. Three patients with PCD had CBF values above this threshold; however, two of them exhibited abnormal CBP. Sample type, patient age, and the presence of airway pathogens did not significantly influence CBF measurements. Conclusions: CBF and CBP analysis using DHSV represents a useful first-line screening tool within a multifaceted diagnostic approach for PCD, allowing rapid identification of patients who should undergo further confirmatory testing.

Fields of Study & Subjects

Cystic Fibrosis Research Advances Genetic and Kidney Cyst Diseases Immunodeficiency and Autoimmune Disorders

Publication details

Journal / source
Diagnostics
Volume
16
Issue
5
Pages
704–704
Publication date
27 February 2026
Language
EN