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. 2018 Feb 15;14(2):245-252.
doi: 10.5664/jcsm.6944.

Nocturnal REM Sleep Without Atonia Is a Diagnostic Biomarker of Pediatric Narcolepsy

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Free PMC article

Nocturnal REM Sleep Without Atonia Is a Diagnostic Biomarker of Pediatric Narcolepsy

Saadoun Bin-Hasan et al. J Clin Sleep Med. .
Free PMC article

Abstract

Study objectives: Compare nocturnal REM sleep without atonia (nRWA) and REM sleep behavior disorder (RBD) between pediatric patients with and without narcolepsy and determine if the nRWA index is a valid diagnostic biomarker for narcolepsy.

Methods: Retrospective cohort study of children ages 6 to 18 years who completed a nocturnal polysomnogram (PSG) and Multiple Sleep Latency Test (MSLT). Our study sample included 11 patients with narcolepsy type 1 (NT1), 6 with narcolepsy type 2 (NT2), 12 with idiopathic hypersomnia (IH), and 11 with subjective hypersomnia (sHS). We compared group nRWA indices (epochs of RWA/total stage R sleep epochs) from the nocturnal PSGs and analyzed nRWA index receiver operating curve (ROC) statistics for narcolepsy diagnosis.

Results: The median nRWA index of patients with NT1 was 15 to 30 times higher compared to sHS and IH (Ps < .005) but similar to that of the NT2 group (P = .46). RBD was present in 25% of patients with narcolepsy (NT1 and NT2). In comparing those with and without narcolepsy, the nRWA index area under the curve was 0.87 (0.6), 95% confidence interval (CI) = 0.75 to 0.99, P < .001. The threshold of having ≥ 1% of stage R sleep epochs with nRWA yielded a sensitivity of 88.2%, 95% CI = 63.6-98.5 and specificity of 60.9%, 95% CI = 38.5 to 80.3 for diagnosis of narcolepsy. In contrast, a threshold of ≥ 8% yielded a specificity of 95.7%, 95% CI = 78.1 to 99.9 and sensitivity of 52.9%, 95% CI = 27.8 to 77.

Conclusions: The nRWA index is a very good diagnostic biomarker of pediatric narcolepsy. Depending on the clinical cutoffs utilized, this biomarker can identify more children/adolescents with narcolepsy using just the PSG or reduce false-positive diagnostic results.

Keywords: REM sleep behavior disorder; REM sleep without atonia; children; diagnosis; idiopathic hypersomnia; narcolepsy; rapid eye movement sleep.

Figures

Figure 1
Figure 1. Group comparison of nRWA index on the PSG.
Median nRWA indices are plotted with bar representing median values and whiskers representing minimum and maximum values. Outliers (> 3 standard deviations from median) are designated as circles. IH = idiopathic hypersomnia, nRWA = nocturnal REM sleep without atonia, NT1 = narcolepsy type 1, NT2 = narcolepsy type 2, PSG = polysomnography, sHS = subjective hypersomnia.
Figure 2
Figure 2. ROC of nRWA index for diagnosis of pediatric narcolepsy.
Diagonal segments are produced by ties. ROC curve for diagnosis of pediatric narcolepsy (NT1 and NT2) compared to those without narcolepsy (IH and sHS): AUC = 0.87 (0.6), 95% CI = 0.75–0.99, P < .001. AUC = area under the curve, CI = confidence interval, IH = idiopathic hypersomnia, nRWA = nocturnal REM sleep without atonia, NT1 = narcolepsy type 1, NT2 = narcolepsy type 2, ROC = receiver operating characteristic, sHS = subjective hypersomnia.

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