Nocturnal rapid eye movement sleep latency for identifying patients with narcolepsy/hypocretin deficiency.
Nocturnal rapid eye movement sleep latency for identifying patients with narcolepsy/hypocretin deficiency.
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DOI：
10.1001/jamaneurol.2013.1589
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发表时间：
2013-07
期刊：
影响因子：
29
通讯作者：
Mignot, Emmanuel
中科院分区：
文献类型：
--
作者：
Andlauer, Olivier;Moore, Hyatt;Jouhier, Laura;Drake, Christopher;Peppard, Paul E.;Han, Fang;Hong, Seung-Chul;Poli, Francesca;Plazzi, Giuseppe;O'Hara, Ruth;Haffen, Emmanuel;Roth, Thomas;Young, Terry;Mignot, Emmanuel
发作性睡病是一种与HLA-DQB 1 *06：02相关的疾病，由下丘脑泌素（食欲素）缺乏引起，使用夜间多导睡眠图（NPSG）后的多次睡眠潜伏期测试（MPEST）进行诊断。在许多患者中，在NPSG期间也观察到短的快速眼动睡眠潜伏期（REML），但未用于诊断。确定夜间REML测量对发作性睡病/下丘脑分泌素缺乏症的诊断准确性和临床实用性。使用NPSG REML和MPEST结果的受试者工作特征曲线的观察性研究（1976年5月至2011年9月在美国、中国、韩国和欧洲的大学医学中心进行的睡眠研究），以确定与不同样本相比的嗜睡症/下丘脑泌素缺乏症的最佳诊断临界值：对照组、患有其他睡眠障碍的患者、患有其他睡眠过度的患者和具有正常下丘脑泌素水平的发作性睡病患者。进行了越来越严格的比较。在第一次比较中，从1749名患者中选择了516名年龄和性别匹配的发作性睡病/下丘脑泌素缺乏症患者，并与516名对照组进行了比较。在第二项比较中，749名连续患者接受任何睡眠障碍的睡眠评估（发作性睡病的预测概率较低），通过最终诊断发作性睡病/下丘脑泌素缺乏症进行组内比较。在第三次比较中，254例具有较高的发作性睡病预测概率的患者通过其最终诊断在组内进行比较。最后，将118例发作性睡病/下丘脑泌素缺乏症患者与118例年龄和性别匹配的诊断为发作性睡病但下丘脑泌素水平正常的患者进行比较。NPSG REML和MPEG4作为发作性睡病/下丘脑泌素缺乏症诊断试验的敏感性和特异性该诊断定义为发作性睡病伴卡他汀+HLA-DQB 1 *06：02阳性（无可用的脑脊液下丘脑泌素-1结果）或发作性睡病伴记录的低（≤110 pg/mL）脑脊液下丘脑泌素-1水平。短REML NPSG期间（≤15分钟）具有高度特异性（99.2% [95% CI，98.5%-100.0%]的516例和99.6% [95% CI，99.1%-100.0%]的735例）但不敏感（50.6% [95% CI，46.3%-54.9%]/516和35.7% [95% CI，10.6%-60.8%]/14），发作性睡病/下丘脑泌素缺乏症患者vs基于人群的对照或接受夜间睡眠研究的所有睡眠障碍患者（曲线下面积分别为0.799 [95% CI，0.771-0.826]和0.704 [95% CI，0.524-0.907]）。在患有中枢性睡眠过度的患者中，因此发作性睡病的预测试概率较高，短REML仍然具有高度特异性（95.4% [95% CI，90.4%-98.3%]/132）且敏感性相似（57.4% [95% CI，48.1%-66.3%]/122）（曲线下面积，0.765 [95% CI，0.707-0.831]）。该高预试概率样本的阳性预测值为92.1%（95% CI，83.6%-97.0%）。在评估可能的发作性睡病的患者中，NPSG的短REML（≤15分钟）具有高特异性和阳性预测值，可被视为诊断性，而无需使用MPERT;然而，缺乏短REML需要随后的MPERT。
Narcolepsy, a disorder associated with HLA-DQB1*06:02 and caused by hypocretin (orexin) deficiency, is diagnosed using the Multiple Sleep Latency Test (MSLT) following nocturnal polysomnography (NPSG). In many patients, a short rapid eye movement sleep latency (REML) during the NPSG is also observed but not used diagnostically. To determine diagnostic accuracy and clinical utility of nocturnal REML measures in narcolepsy/hypocretin deficiency. Observational study using receiver operating characteristic curves for NPSG REML and MSLT findings (sleep studies performed between May 1976 and September 2011 at university medical centers in the United States, China, Korea, and Europe) to determine optimal diagnostic cutoffs for narcolepsy/hypocretin deficiency compared with different samples: controls, patients with other sleep disorders, patients with other hypersomnias, and patients with narcolepsy with normal hypocretin levels. Increasingly stringent comparisons were made. In a first comparison, 516 age- and sex-matched patients with narcolepsy/hypocretin deficiency were selected from 1749 patients and compared with 516 controls. In a second comparison, 749 successive patients undergoing sleep evaluation for any sleep disorders (low pretest probability for narcolepsy) were compared within groups by final diagnosis of narcolepsy/hypocretin deficiency. In the third comparison, 254 patients with a high pretest probability of having narcolepsy were compared within group by their final diagnosis. Finally, 118 patients with narcolepsy/hypocretin deficiency were compared with 118 age- and sex-matched patients with a diagnosis of narcolepsy but with normal hypocretin levels. Sensitivity and specificity of NPSG REML and MSLT as diagnostic tests for narcolepsy/hypocretin deficiency. This diagnosis was defined as narcolepsy associated with cataplexy plus HLA-DQB1*06:02 positivity (no cerebrospinal fluid hypocretin-1 results available) or narcolepsy with documented low (≤110 pg/mL) cerebrospinal fluid hypocretin-1 level. Short REML (≤15 minutes) during NPSG was highly specific (99.2% [95% CI, 98.5%–100.0%] of 516 and 99.6% [95% CI, 99.1%–100.0%] of 735) but not sensitive (50.6% [95% CI, 46.3%–54.9%] of 516 and 35.7% [95% CI, 10.6%–60.8%] of 14) for patients with narcolepsy/hypocretin deficiency vs population-based controls or all patients with sleep disorders undergoing a nocturnal sleep study (area under the curve, 0.799 [95% CI, 0.771–0.826] and 0.704 [95% CI, 0.524–0.907], respectively). In patients with central hypersomnia and thus a high pretest probability for narcolepsy, short REML remained highly specific (95.4% [95% CI, 90.4%–98.3%] of 132) and similarly sensitive (57.4% [95% CI, 48.1%–66.3%] of 122) for narcolepsy/hypocretin deficiency (area under the curve, 0.765 [95% CI, 0.707–0.831]). Positive predictive value in this high pretest probability sample was 92.1% (95% CI, 83.6%–97.0%). Among patients being evaluated for possible narcolepsy, short REML (≤15 minutes) at NPSG had high specificity and positive predictive value and may be considered diagnostic without the use of an MSLT; absence of short REML, however, requires a subsequent MSLT.
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影响因子：
30.8
作者：
Hallmayer, Joachim;Faraco, Juliette;Mignot, Emmanuel
通讯作者：
Mignot, Emmanuel
影响因子：
10.6
作者：
Johnson, EO;Breslau, N;Rosenthal, L
通讯作者：
Rosenthal, L
影响因子：
9.9
作者：
Dauvilliers, Y;Gosselin, A;Montplaisir, J
通讯作者：
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影响因子：
4.4
作者：
Khalsa, SBS;Conroy, DA;Dijk, DJ
通讯作者：
Dijk, DJ
影响因子：
4.4
作者：
Edinger, JD;Glenn, DM;Meeks, G
通讯作者：
Meeks, G