Tidal breathing nasal nitric oxide measurement as a test for primary ciliary dyskinesia in young children

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Abstract
Background: Nasal nitric oxide (nNO) measurement during velum closure is a useful test for primary ciliary dyskinesia (PCD) but is not feasible in young children. Measurement during tidal breathing is an alternative approach. Our objectives were to define a protocol with age-specific cut-off nNO values during tidal breathing and evaluate this method as an adjunctive test for PCD. Methods: A standardised method for nNO measurement during tidal breathing was developed and tested at the University of North Carolina in children aged −1 at 2 months to 47.7 nL·min −1 at 5 years of age. Receiver operating characteristic curves had high areas under each curve for nNO cut-offs, ranging from 0.98 to 0.99 across the ages represented in the study. Validation testing at participating sites showed tidal breathing nNO accurately identified 93% of the 60 children aged <6 years with PCD confirmed by ultrastructural ciliary analysis or genetic testing, with high specificity. Conclusion: Tidal breathing nNO measurement is a useful adjunctive test to distinguish young children with PCD from healthy controls as young as 2 months using age-specific cut-off nNO values.
Funding Information
  • National Center for Advancing Translational Sciences (UM1 TR004399)
  • National Heart, Lung, and Blood Institute (R01HL071798)
  • National Heart, Lung, and Blood Institute (U54HL096458)

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