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Chest. 2019 Feb 28. pii: S0012-3692(19)30205-3. doi: 10.1016/j.chest.2019.01.036. [Epub ahead of print]

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

Author information

1
Primary Ciliary Dyskinesia Centre, NIHR Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK; Academic Unit of Clinical and Experimental Medicine, University of Southampton Faculty of Medicine, Southampton, UK.
2
Primary Ciliary Dyskinesia Centre, Paediatric Respiratory Medicine, Royal Brompton Hospital, London UK; Division of Molecular and Clinical Medicine, Scottish Centre for Respiratory Research, University of Dundee, Dundee, UK.
3
Centre for PCD Diagnosis and Research, Department of Infection, Immunity and Inflammation, University of Leicester, and Robert Kilpatrick Clinical Sciences Building, Leicester Royal Infirmary, Leicester, UK.
4
Primary Ciliary Dyskinesia Centre, Department of Paediatrics and Department of Paediatric Respiratory Medicine, Imperial College and Royal Brompton Hospital, London UK.
5
PCD Family Support Group, Milton Keynes, UK.
6
Centre for PCD Diagnosis and Research, Department of Infection, Immunity and Inflammation, University of Leicester, and Robert Kilpatrick Clinical Sciences Building, Leicester Royal Infirmary, Leicester, UK; Infection, Immunity, Inflammation and Physiological Medicine Programme, Institute of Child Health, University College London, London, UK.
7
Research Design Service South Central, National Institute for Health Research, and University Hospital Southampton NHS Foundation Trust, Southampton, UK.
8
Primary Ciliary Dyskinesia Centre, NIHR Biomedical Research Centre, University Hospital Southampton NHS Foundation Trust, Southampton, UK; Academic Unit of Clinical and Experimental Medicine, University of Southampton Faculty of Medicine, Southampton, UK. Electronic address: jlucas1@soton.ac.uk.

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.

KEYWORDS:

accuracy; diagnostic tests; microscopy; primary ciliary dyskinesia; sensitivity and specificity

PMID:
30826306
DOI:
10.1016/j.chest.2019.01.036
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