Ouyang Hui, Han Fang, Zhou Ze-Chen, Zhang Jun
Department of Clinical Neurology, Peking University People's Hospital, Beijing, China.
Department of Clinical Pulmonology, Peking University People's Hospital, Beijing, China.
Neural Regen Res. 2020 Oct;15(10):1887-1893. doi: 10.4103/1673-5374.280322.
Early- and late-onset narcolepsy constitutes two distinct diagnostic subgroups. However, it is not clear whether symptomology and genetic risk factors differ between early- and late-onset narcoleptics. This study compared clinical data and single-nucleotide polymorphisms (SNPs) between early- and late-onset patients in a large cohort of 899 Han Chinese narcolepsy patients. Blood, cerebrospinal fluid, and clinical data were prospectively collected from patients, and patients were genotyped for 40 previously reported narcolepsy risk-conferring SNPs. Genetic risk scores (GRSs), associations of five different sets of SNPs (GRS1-GRS5) with early- and late-onset narcolepsy, were evaluated using logistic regression and receiver operating characteristic curves. Mean sleep latency was significantly shorter in early-onset cases than in late-onset cases. Symptom severity was greater among late-onset patients, with higher rates of sleep paralysis, hypnagogic hallucinations, health-related quality of life impairment, and concurrent presentation with four or more symptoms. Hypocretin levels did not differ significantly between early- and late-onset cases. Only rs3181077 (CCR1/CCR3) and rs9274477 (HLA-DQB1) were more prevalent among early-onset cases. Only GRS1 (26 SNPs; OR = 1.513, 95% CI: 0.893-2.585; P < 0.05) and GRS5 (6 SNPs; OR = 1.893, 95% CI: 1.204-2.993; P < 0.05) were associated with early-onset narcolepsy, with areas under the receiver operating characteristic curves of 0.731 and 0.732, respectively. Neither GRS1 nor GRS5 included SNPs in HLA regions. Our results indicate that symptomology and genetic risk factors differ between early- and late-onset narcolepsy. This protocol was approved by the Institutional Review Board (IRB) Panels on Medical Human Subjects at Peking University People's Hospital, China (approval No. Yuanlunshenlinyi 86) in October 2011.
早发型发作性睡病和晚发型发作性睡病构成两个不同的诊断亚组。然而,早发型和晚发型发作性睡病患者的症状学和遗传风险因素是否存在差异尚不清楚。本研究比较了899例中国汉族发作性睡病患者队列中早发型和晚发型患者的临床数据和单核苷酸多态性(SNP)。前瞻性收集患者的血液、脑脊液和临床数据,并对患者进行40个先前报道的发作性睡病风险SNP的基因分型。使用逻辑回归和受试者工作特征曲线评估遗传风险评分(GRS),即五组不同SNP(GRS1-GRS5)与早发型和晚发型发作性睡病的关联。早发型患者的平均睡眠潜伏期显著短于晚发型患者。晚发型患者的症状严重程度更高,睡眠麻痹、入睡前幻觉、健康相关生活质量受损以及同时出现四种或更多症状的发生率更高。早发型和晚发型患者的下丘脑分泌素水平无显著差异。仅rs3181077(CCR1/CCR3)和rs9274477(HLA-DQB1)在早发型患者中更为常见。仅GRS1(26个SNP;OR = 1.513,95%CI:0.893-2.585;P < 0.05)和GRS5(6个SNP;OR = 1.893,95%CI:1.204-2.993;P < 0.05)与早发型发作性睡病相关,受试者工作特征曲线下面积分别为0.731和0.732。GRS1和GRS5均不包括HLA区域的SNP。我们的结果表明,早发型和晚发型发作性睡病的症状学和遗传风险因素存在差异。本研究方案于2011年10月获得中国北京大学人民医院医学人体研究机构审查委员会(IRB)小组批准(批准号:Yuanlunshenlinyi 86)。