» Articles » PMID: 37701364

Quantitative Tc-albumin Colloid Nasal Mucociliary Clearance As an Outcome in Primary Ciliary Dyskinesia

Overview
Journal ERJ Open Res
Specialty Pulmonary Medicine
Date 2023 Sep 13
PMID 37701364
Authors
Affiliations
Soon will be listed here.
Abstract

Background: Primary ciliary dyskinesia (PCD) is an inherited disorder in which dyskinetic cilia cause impaired mucociliary clearance of upper and lower airways. Airway ciliary movement can be indirectly tested after administration of a radiolabelled tracer to the lower airways for assessment of pulmonary mucociliary clearance or to the nose for assessing nasal mucociliary clearance (NMC). With this study, we investigated NMC as a quantifiable study outcome parameter in patients with PCD.

Material And Methods: This single centre proof-of-concept study on NMC velocity investigated patients with PCD across different genotypes and nasal nitric oxide (nasal NO) levels. Healthy controls were used for comparison. NMC was determined as velocity in mm·min of a nasally applied Tc-albumin colloid tracer. Using a gamma camera, repeated dynamic series of images each lasting 30 s were acquired during a 10-minute period and digitally stored.

Results: NMC velocity was investigated in seven patients with PCD (aged 9-31 years) and five adult healthy controls. Mean NMC velocity in healthy controls (8.5 mm·min) was significantly higher compared with people with PCD (0.00 mm·min, p<0.0001). NMC was completely absent in all included patients with PCD across different PCD genotypes and regardless of nasal NO values. The success rate of the test was 100% in both groups.

Conclusion: NMC velocity discriminated highly significantly between patients with PCD and healthy controls. We suggest here a fast and feasible set up for NMC measurements that is easily applicable for any clinical trial involving PCD medication aimed for the nasal compartment, a step before or parallel to conducting clinical trials investigating whole-lung ciliary function in PCD.

Citing Articles

Pulmonary radioaerosol mucociliary clearance assessment: searching for genotype-specific differences and potential as an outcome measure in primary ciliary dyskinesia.

Marthin J, Holgersen M, Nielsen K, Mortensen J ERJ Open Res. 2023; 9(6).

PMID: 38076675 PMC: 10702481. DOI: 10.1183/23120541.00685-2023.

References
1.
Lindberg S, Cervin A, Runer T . Low levels of nasal nitric oxide (NO) correlate to impaired mucociliary function in the upper airways. Acta Otolaryngol. 1997; 117(5):728-34. DOI: 10.3109/00016489709113468. View

2.
Walker W, Young A, Bennett M, Guy M, Carroll M, Fleming J . Pulmonary radioaerosol mucociliary clearance in primary ciliary dyskinesia. Eur Respir J. 2014; 44(2):533-5. DOI: 10.1183/09031936.00011814. View

3.
Donaldson S, Laube B, Mogayzel P, Corcoran T, Pilewski J, Ceppe A . Effect of lumacaftor-ivacaftor on mucociliary clearance and clinical outcomes in cystic fibrosis: Results from the PROSPECT MCC sub-study. J Cyst Fibros. 2021; 21(1):143-145. PMC: 8630086. DOI: 10.1016/j.jcf.2021.05.004. View

4.
De Boeck K, Proesmans M, Mortelmans L, Van Billoen B, Willems T, Jorissen M . Mucociliary transport using 99mTc-albumin colloid: a reliable screening test for primary ciliary dyskinesia. Thorax. 2005; 60(5):414-7. PMC: 1758893. DOI: 10.1136/thx.2004.027680. View

5.
Paff T, Omran H, Nielsen K, Haarman E . Current and Future Treatments in Primary Ciliary Dyskinesia. Int J Mol Sci. 2021; 22(18). PMC: 8470068. DOI: 10.3390/ijms22189834. View