Nalbantoğlu Mecbure, Benbir Gülçin, Karadeniz Derya, Altintaş Ayşe, Savran Oğuz Fatma
Sleep Disorders Unit, Department of Neurology, Cerrahpaşa İstanbul University, İstanbul Faculty of Medicine, İstanbul, Turkey.
Department of Medical Biology, İstanbul University İstanbul Faculty of Medicine, İstanbul, Turkey.
Noro Psikiyatr Ars. 2014 Sep;51(3):283-287. doi: 10.4274/npa.y7051. Epub 2014 Sep 1.
Narcolepsy-Cataplexy syndrome is a rare sleep disorder related with human leukocyte antigens (HLA-DQB10602) caused by the loss of hypothalamic hypocretin/orexin producing neurons. Recently, in European countries, Narcolepsy-Cataplexy syndrome developing following H1N1 vaccination has attracted attention. Our first patient was a 9-year-old boy, who was referred to our clinic with the complaint of daytime sleepiness developed 1.5 month after H1N1 vaccination. After a couple of weeks, weakness of the upper extremities while laughing was added to the clinical picture. He also developed hypnopompic hallucinations and nightmares. Multiple sleep latency test (MSLT) following full-night polysomnography (PSG) showed that the mean sleep latency was 0.6 minutes; and all of the naps had sleep-onset REM periods. Our second patient was a 50 year-oldman, who presented to our clinic complaining of sleep paralysis and REM sleep behavior disorder developed 4 months after H1N1 vaccination. He developed daytime sleepiness 6 months after and cataplexy 8 months after H1N1 vaccination. He was also diagnosed as having Narcolepsy-Cataplexy syndrome upon PSG and MSLT. We investigated HLA-DRB1/ DQB1 locus with Polymerase Chain Reaction-Sequence Specific Primer technique. The first patient had HLA-DQB10602.47 and DQB103.01 heterozygous loci; and the second patient had HLA-DQB10602.47 and DQB1*02.01 heterozygous loci. These patients are the first reported cases of Narcolepsy-Cataplexy syndrome related with H1N1 vaccination in Turkey. Although there is no specific marker, temporal relationship between vaccination and onset of disease symptoms suggests a possible causal relationship. In the presence of an underlying genetic predisposition, it was thought that H1N1 vaccination could trigger Narcolepsy-Cataplexy syndrome.
发作性睡病 - 猝倒综合征是一种罕见的睡眠障碍,与人类白细胞抗原(HLA - DQB10602)相关,由下丘脑分泌食欲素的神经元缺失所致。最近,在欧洲国家,甲型H1N1流感疫苗接种后发生的发作性睡病 - 猝倒综合征引起了关注。我们的首例患者是一名9岁男孩,因接种甲型H1N1流感疫苗1.5个月后出现日间嗜睡症状而转诊至我们诊所。几周后,临床症状中又增加了大笑时上肢无力的症状。他还出现了醒前幻觉和噩梦。整夜多导睡眠图(PSG)检查后的多次睡眠潜伏期试验(MSLT)显示,平均睡眠潜伏期为0.6分钟;所有小睡均出现睡眠起始快速眼动期。我们的第二例患者是一名50岁男性,因接种甲型H1N1流感疫苗4个月后出现睡眠瘫痪和快速眼动睡眠行为障碍而前来我们诊所就诊。接种甲型H1N1流感疫苗6个月后他出现日间嗜睡,8个月后出现猝倒。经PSG和MSLT检查,他也被诊断为发作性睡病 - 猝倒综合征。我们采用聚合酶链反应 - 序列特异性引物技术对HLA - DRB1 / DQB1基因座进行了检测。首例患者具有HLA - DQB10602.47和DQB103.01杂合基因座;第二例患者具有HLA - DQB10602.47和DQB1*02.01杂合基因座。这些患者是土耳其首例报告的与甲型H1N1流感疫苗接种相关的发作性睡病 - 猝倒综合征病例。尽管没有特异性标志物,但疫苗接种与疾病症状发作之间的时间关系提示可能存在因果关系。在存在潜在遗传易感性的情况下,认为甲型H1N1流感疫苗接种可能引发发作性睡病 - 猝倒综合征。