The authors wish to declare that they have no competing interests.
PK performed the search and extraction of information from the papers, organized the material and prepared the first draft of the manuscript. SIP provided advice on the methodology of the search and selection of the papers, contributed to the interpretation of the findings, critically revised the manuscript and contributed towards the final version of the manuscript. PKY conceived the hypothesis of the manuscript, coordinated the whole work, contributed to the interpretation of the findings and contributed intellectually towards the final version of the manuscript. All authors read and approved the final manuscript.
S.I.P. is the main advisor of P.K for his doctoral studies. PK was supported by the European Union’s Seventh Framework Program EC-GA No. 305404 BESTCILIA. The sponsors had no role or involvement in study design; in the collection, analysis and interpretation of data; in the writing of the report; and in the decision to submit the article for publication.
To date, diagnosis of Primary Ciliary Dyskinesia (PCD) remains difficult and challenging. We systematically evaluated the diagnostic performance of nasal Nitric Oxide (nNO) measurement for the detection of PCD, using either velum-closure (VC) or non-velum-closure (non-VC) techniques.
All major electronic databases were searched from inception until March 2015 using appropriate terms. The sensitivity and specificity of nNO measurement was calculated in PCD patients diagnosed by transmission electron microscopy, high speed video-microscopy or genetic testing. Summary receiver operating characteristic (HSROC) curves were drawn using the parameters of the fitted models.
Twelve studies provided data for 13 different populations, including nine case–control (n = 793) and four prospective cohorts (n = 392). The overall sensitivity of nNO measured by VC techniques was 0.95 (95 % CI 0.91–0.97), while specificity was 0.94 (95 % CI 0.88–0.97). The positive likelihood ratio (LR+) of the test was 15.8 (95 % CI 8.1–30.6), whereas the negative likelihood ratio (LR-) was 0.06 (95 % CI 0.04–0.09). For non-VC techniques, the overall sensitivity of nNO measurement was 0.93 (95 % CI 0.89–0.96) whereas specificity was 0.95 (95 % CI 0.82–0.99). The LR+ of the test was 18.5 (95 % CI 4.6–73.8) whereas the LR- was 0.07 (95 % CI 0.04–0.12).
Diagnostic accuracy of nNO measurement both with VC and non-VC maneuvers is high and can be effectively employed in the clinical setting to detect PCD even in young children, thus potentiating early diagnosis. Measurement of nNO merits to be part of a revised diagnostic algorithm with the most efficacious combination of tests to achieve PCD diagnosis.
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Montella S, Alving K, De Stefano S, Di Micco LL, Di Giorgio A, Santamaria F. Nasal nitric oxide measurement using continuous aspiration by hand-held device discriminates patients with primary ciliary dyskinesia from healthy subjects. Eur Respir J. 2012;40 Suppl 56:3338.
Santamaria F, De Stefano S, Montella S, Barbarano F, Iacotucci P, Ciccarelli R, et al. Nasal nitric oxide assessment in primary ciliary dyskinesia using aspiration, exhalation, and humming. Med Sci Monit. 2008;14(2):CR80–5. PubMed
Moreno Galdo A, Vizmanos Lamotte G, Reverte Bover C, Gartner S, Cobos Barroso N, Rovira Amigo S, et al. Value of nasal nitric oxide in the diagnosis of primary ciliary dyskinesia. An Pediatr (Barc). 2010;73(2):88–93. CrossRef
- Diagnostic accuracy of nasal nitric oxide for establishing diagnosis of primary ciliary dyskinesia: a meta-analysis
Stefania I. Papatheodorou
Panayiotis K. Yiallouros
- BioMed Central
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