Neurology Department, Hôpital d'Instruction des Armées Percy, Clamart, France.
Sleep Disorders Unit, Hôpital Pitié-Salpêtrière, Paris, France.
J Clin Sleep Med. 2023 Apr 1;19(4):837-841. doi: 10.5664/jcsm.10448.
Recent studies suggest that sleep disorders are present in two-thirds of patients with autoimmune encephalitis. In anti-Ma2 encephalitis, hypersomnia appears to be frequent. However, only few cases of type 1 narcolepsy have been reported to date with anti-Ma2 encephalitis. We report 2 new cases of patients with narcolepsy secondary to anti-Ma2 encephalitis. Patient 1, a 68-year-old man, had narcolepsy type 1, including sleep attacks, cataplexy, abnormal Multiple Sleep Latency Tests and hypocretin-1 deficiency (< 50 ng/L) in the cerebrospinal fluid (CSF), associated with a cerebellar syndrome. Anti-Ma2 antibodies were present in the serum and CSF and antivoltage-gated potassium channel antibodies in the serum. He benefited from a treatment with pitolisant. Patient 2, a 42-year-old man, had narcolepsy type 2, including hypersomnolence, no cataplexy, intermediate CSF levels of hypocretin-1 (138 ng/L), abnormal Multiple Sleep Latency Tests, and a limbic encephalitis presentation. Anti-Ma2 antibodies were present in the serum and CSF, and anti-Ma1 antibodies were in the CSF. For both, repeated polysomnographies were necessary to establish the precise diagnosis of central hypersomnia, emphasizing the importance of carrying out sleep investigations in a tertiary neurology center with sleep medicine expertise in patients with anti-Ma2 encephalitis.
Brunet de Courssou J-B, Testard P, Sallansonnet-Froment M, et al. Narcolepsy secondary to anti-Ma2 encephalitis: two case reports. . 2023;19(4):837-841.
最近的研究表明,三分之二的自身免疫性脑炎患者存在睡眠障碍。在抗 Ma2 脑炎中,嗜睡似乎很常见。然而,迄今为止,只有少数抗 Ma2 脑炎合并 1 型发作性睡病的病例报告。我们报告了 2 例新的抗 Ma2 脑炎继发发作性睡病患者。患者 1 为 68 岁男性,患有 1 型发作性睡病,包括睡眠发作、猝倒、异常多次睡眠潜伏期试验和脑脊液中下丘脑泌素-1 缺乏症(<50ng/L),伴有小脑综合征。血清和脑脊液中存在抗 Ma2 抗体,血清中存在抗电压门控钾通道抗体。他受益于吡唑酰胺治疗。患者 2 为 42 岁男性,患有 2 型发作性睡病,包括嗜睡、无猝倒、脑脊液中下丘脑泌素-1 水平中等(138ng/L)、异常多次睡眠潜伏期试验和边缘性脑炎表现。血清和脑脊液中存在抗 Ma2 抗体,脑脊液中存在抗 Ma1 抗体。两者均需要重复多导睡眠图以明确中枢性嗜睡的诊断,这强调了在具有睡眠医学专业知识的三级神经病学中心对抗 Ma2 脑炎患者进行睡眠研究的重要性。
Brunet de Courssou J-B,Testard P,Sallansonnet-Froment M,等。抗 Ma2 脑炎继发发作性睡病:两例报告。. 2023;19(4):837-841。