University of Toronto, Toronto, Ontario, Canada.
Division of Respiratory Medicine, Department of Pediatrics, Hospital for Sick Children, Toronto, Ontario, Canada.
J Clin Sleep Med. 2018 Feb 15;14(2):245-252. doi: 10.5664/jcsm.6944.
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.
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.
The median nRWA index of patients with NT1 was 15 to 30 times higher compared to sHS and IH (s < .005) but similar to that of the NT2 group ( = .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, < .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.
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.
比较伴有和不伴有嗜睡症的儿科患者的夜间 REM 睡眠无动（nRWA）和 REM 睡眠行为障碍（RBD），并确定 nRWA 指数是否是嗜睡症的有效诊断生物标志物。
对年龄在 6 至 18 岁之间完成夜间多导睡眠图（PSG）和多次睡眠潜伏期试验（MSLT）的儿童进行回顾性队列研究。我们的研究样本包括 11 例 1 型发作性睡病（NT1）患者、6 例 2 型发作性睡病（NT2）患者、12 例特发性嗜睡症（IH）患者和 11 例主观性嗜睡症（sHS）患者。我们比较了夜间 PSG 中各组的 nRWA 指数（RWA 期/总 R 期睡眠期），并分析了 nRWA 指数对嗜睡症诊断的受试者工作特征曲线（ROC）统计。
NT1 患者的 nRWA 指数中位数比 sHS 和 IH 高 15 至 30 倍（s <.005），但与 NT2 组相似（=.46）。25%的嗜睡症患者（NT1 和 NT2）存在 RBD。在比较有和无嗜睡症的患者时，nRWA 指数曲线下面积为 0.87（0.6），95%置信区间（CI）为 0.75 至 0.99，<.001。nRWA 指数≥ 1%的 R 期睡眠期具有诊断嗜睡症的敏感性为 88.2%，95%CI=63.6-98.5，特异性为 60.9%，95%CI=38.5 至 80.3。相比之下，≥ 8%的阈值特异性为 95.7%，95%CI=78.1 至 99.9，敏感性为 52.9%，95%CI=27.8 至 77.
nRWA 指数是小儿嗜睡症的非常好的诊断生物标志物。根据使用的临床截止值，该生物标志物可以仅使用 PSG 识别更多的嗜睡症儿童/青少年，或者减少假阳性诊断结果。
