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Accuracy of High-Speed Video Analysis to Diagnose Primary Ciliary Dyskinesia

Rubbo, B; Shoemark, A; Jackson, CL; Hirst, R; Thompson, J; Hayes, J; Frost, E; ... Lucas, JS; + view all (2019) Accuracy of High-Speed Video Analysis to Diagnose Primary Ciliary Dyskinesia. Chest , 155 (5) pp. 1008-1017. 10.1016/j.chest.2019.01.036. Green open access

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Abstract

Background Diagnosis of primary ciliary dyskinesia (PCD) relies on a combination of tests. High-speed video microscopy analysis (HSVA) is widely used to contribute to the diagnosis. It can be analyzed on the day of diagnostic consultation, but the qualitative analyses are subjective. Diagnostic accuracy and reliability of assessing ciliary function have not been robustly evaluated. We aimed to establish the accuracy of HSVA to diagnose PCD compared with a combination of tests, and to assess the interobserver reliability of HSVA analysis. Methods We randomly selected and anonymized archived videos from 120 patients seen at three UK PCD centers. Three experienced scientists independently reviewed six videos per patient, using a standardized proforma, blinded to diagnostic and clinical data. We compared study outcomes with two references: (1) a combination of diagnostic tests in accordance with the European Respiratory Society PCD diagnostic guidelines and (2) original clinical outcome determined by all available diagnostic tests. Results HSVA had excellent sensitivity and specificity to diagnose PCD: (1) 100% and 96%, respectively, compared with ERS guidelines, and (2) 96% and 91% compared with diagnostic outcomes. There was high interobserver agreement for “PCD-positive” outcomes (κ = 0.7). Conclusions Specialist scientists accurately diagnosed PCD using HSVA, with high interobserver agreement. HSVA can be reliably used to counsel patients and commence treatment on the day of testing while awaiting confirmatory investigations.

Type: Article
Title: Accuracy of High-Speed Video Analysis to Diagnose Primary Ciliary Dyskinesia
Open access status: An open access version is available from UCL Discovery
DOI: 10.1016/j.chest.2019.01.036
Publisher version: https://doi.org/10.1016/j.chest.2019.01.036
Language: English
Additional information: This is an open access article under the CC BY-NC-ND license (http:// creativecommons.org/licenses/by-nc-nd/4.0/).
Keywords: accuracy; diagnostic tests; microscopy; primary ciliary dyskinesia; sensitivity and specificity
UCL classification: UCL
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Infection, Immunity and Inflammation Dept
URI: https://discovery.ucl.ac.uk/id/eprint/10083861
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