Erkent Irem, Elibol Bulent, Saka Esen, Saygi Serap, Tezer Irsel
Department of Neurology, Faculty of Medicine, Hacettepe University, Ankara, Turkey.
Neurol Sci. 2023 Apr;44(4):1351-1360. doi: 10.1007/s10072-022-06513-x. Epub 2022 Dec 1.
Sleep disorders in patients with autoimmune encephalitis (AE) are increasingly reported. Early recognition and treatment have significant importance regarding the potential of sleep disorders' effect on morbidity and even mortality. There are a limited number of studies related to polysomnography (PSG) in these patients. Here, we report the clinical and PSG data of patients with AE and sleep disorders, with a particular interest in sleep-related breathing disorders (SRBD).
Seventeen patients with diagnosed AE and acute or subacute onset sleep complaints who underwent video-electroencephalography-PSG recordings in our tertiary center were investigated.
The mean age was 50, with eight females and nine males. The detected antibodies were against leucine-rich glioma-inactivated 1(LGI-1) in 6, anti-contactin-associated protein-2(CASPR2) in 3, voltage-gated potassium channel complex antigens(VGKC) in 1, anti-glycine in 1, dipeptidyl-peptidase-like protein-6(DPPX) in 1, anti-Hu in 1, and anti-amphiphysin in 1. All commercially available and known autoimmune encephalitis-related antibodies were negative in 3 of the patients. Final diagnosis after PSG was circadian rhythm sleep disorder (n = 3), periodic limb movement disorder (n = 3), insomnia (n = 5), central apnea with or without Cheyne-Stokes breathing (CSB) (n = 4), obstructive sleep apnea (OSA) (n = 4), non-rapid eye movement (NREM) and REM parasomnia (n = 8), faciobrachial dystonic seizures (n = 2), and subclinical seizures (n = 1). Sleep microstructure was disrupted in 9, REM periods without atonia occurred in 4, and brief sleep fragments consisting of theta activity interspersed with faster rhythms existed in 7 patients. Nearly half of our patients (47%) had SRBD, and the mean apnea-hypopnea index (AHI) was 14.
Sleep disorders are frequent and essential components of AEs. Systematic clinical questionnaires and routine PSG assessments would significantly impact the correct diagnosis and proper treatment of SRBD and the overall prognosis of AE.
自身免疫性脑炎(AE)患者睡眠障碍的报道日益增多。鉴于睡眠障碍对发病率甚至死亡率的潜在影响,早期识别和治疗具有重要意义。关于这些患者多导睡眠图(PSG)的研究数量有限。在此,我们报告AE合并睡眠障碍患者的临床和PSG数据,尤其关注睡眠相关呼吸障碍(SRBD)。
对在我们三级中心接受视频脑电图-PSG记录的17例诊断为AE且有急性或亚急性发作睡眠主诉的患者进行了调查。
平均年龄为50岁,女性8例,男性9例。检测到的抗体中,抗富亮氨酸胶质瘤失活蛋白1(LGI-1)6例,抗接触蛋白相关蛋白2(CASPR2)3例,电压门控钾通道复合抗原(VGKC)1例,抗甘氨酸1例,二肽基肽酶样蛋白6(DPPX)1例,抗Hu 1例,抗 amphiphysin 1例。3例患者所有可商购的已知自身免疫性脑炎相关抗体均为阴性。PSG检查后的最终诊断为昼夜节律睡眠障碍(n = 3)、周期性肢体运动障碍(n = 3)、失眠(n = 5)、伴或不伴陈-施呼吸(CSB)的中枢性呼吸暂停(n = 4)、阻塞性睡眠呼吸暂停(OSA)(n = 4)、非快速眼动(NREM)和快速眼动睡眠行为障碍(n = 8)、面臂肌张力障碍性癫痫(n = 2)以及亚临床癫痫(n = 1)。9例患者睡眠微结构被破坏,4例出现无肌张力的快速眼动期,7例患者存在由θ活动夹杂更快节律组成的短暂睡眠片段。近一半患者(47%)存在SRBD,平均呼吸暂停低通气指数(AHI)为14。
睡眠障碍是AE常见且重要的组成部分。系统的临床问卷和常规PSG评估将对SRBD的正确诊断和恰当治疗以及AE的总体预后产生重大影响。