Diagnostic accuracy of nasal nitric oxide measurement for establishing diagnosis of primary ciliary dyskinesia: A systematic review and meta-analysis

Abstract

Background: Diagnosis of Primary Ciliary Dyskinesia (PCD) remains difficult and challenging. We evaluated the diagnostic performance of nasal Nitric Oxide (nNO) for detection of PCD, using either Velum-Closure (VC) or a non-Velum-Closure (non-VC) technique. Methods: All major electronic databases were searched from inception until September 2014 using appropriate terms. The sensitivity and specificity of nNO measurement was compared against diagnoses of PCD achieved by transmission electron microscopy (TEM) and high speed video-microscopy (HSVM) or genetic testing. We also drew summary receiver operating curves (HSROC) using parameters of the fitted models. Results: Twelve studies were synthesized, nine case-control (n=793) and three prospective cohorts (n=306). The summary sensitivity of low nNO measured by VC techniques was 0.96 (95% CIs 0.90-0.98), while specificity was 0.95 (95% CIs 0.89-0.97) (Figure 1a). For the non-VC techniques, the summary sensitivity of nNO to detect PCD was 0.94 (95% CIs 0.89-0.97) whereas specificity was 0.94 (95% CIs 0.77-0.99) (Figure 1b). Conclusion: Overall diagnostic performance of nNO measurement both with VC and non-VC maneuvers as reported in different studies underlines the effectiveness of nNO against TEM and HSVM or genetic testing to detect PCD, even in young children, thus potentiating early diagnosis and improved prognosis.