Hypersomnia in anti-glutamic acid decarboxylase 65 (GAD65) associated neurological syndromes: A pilot study.
autoimmune diseases of the nervous system
encephalitis
epilepsy
hypersomnia
hypersomnolence
stiff-person syndrome
Journal
European journal of neurology
ISSN: 1468-1331
Titre abrégé: Eur J Neurol
Pays: England
ID NLM: 9506311
Informations de publication
Date de publication:
03 Nov 2023
03 Nov 2023
Historique:
revised:
28
09
2023
received:
10
08
2023
accepted:
18
10
2023
medline:
3
11
2023
pubmed:
3
11
2023
entrez:
3
11
2023
Statut:
aheadofprint
Résumé
Despite their detrimental impact on the quality of life in autoimmune encephalitis, sleep disorders have not been investigated in anti-glutamic acid decarboxylase (GAD65) associated neurological syndromes. Six consecutive adult patients diagnosed with anti-GAD65-associated neurological syndromes (four with limbic encephalitis and two with stiff-person syndrome) and 12 healthy controls were enrolled. Participants underwent sleep interviews and sleep studies including night-time video-polysomnography, followed by five daytime multiple sleep latency tests (MSLTs, to assess propensity to fall asleep) and an 18 h bed rest polysomnography (to assess excessive sleep need). Patients reported the need for daily naps and that their cognition and quality of life were altered by sleepiness, but they had normal scores on the Epworth sleepiness scale. Compared with controls, sleep latencies during the MSLT were shorter in the patient group (median 5.8 min, interquartile range [IQR] 4.5, 6.0 vs. 17.7 min, IQR 16.3, 19.7, p = 0.001), and the arousal index was reduced (2.5/h, IQR 2.3, 3.0 vs. 22.3/h, IQR 13.8, 30.0, p = 0.002), although total sleep time was similar between groups (621 min, IQR 464, 651 vs. 542.5 min, IQR 499, 582, p = 0.51). Remarkably, all six patients had MSLT latencies ≤8 min, indicating severe sleepiness. No parasomnia or sleep-disordered breathing was detected. Central hypersomnia is a relevant characteristic of anti-GAD65-associated neurological syndromes.
Sections du résumé
BACKGROUND AND PURPOSE
OBJECTIVE
Despite their detrimental impact on the quality of life in autoimmune encephalitis, sleep disorders have not been investigated in anti-glutamic acid decarboxylase (GAD65) associated neurological syndromes.
METHODS
METHODS
Six consecutive adult patients diagnosed with anti-GAD65-associated neurological syndromes (four with limbic encephalitis and two with stiff-person syndrome) and 12 healthy controls were enrolled. Participants underwent sleep interviews and sleep studies including night-time video-polysomnography, followed by five daytime multiple sleep latency tests (MSLTs, to assess propensity to fall asleep) and an 18 h bed rest polysomnography (to assess excessive sleep need).
RESULTS
RESULTS
Patients reported the need for daily naps and that their cognition and quality of life were altered by sleepiness, but they had normal scores on the Epworth sleepiness scale. Compared with controls, sleep latencies during the MSLT were shorter in the patient group (median 5.8 min, interquartile range [IQR] 4.5, 6.0 vs. 17.7 min, IQR 16.3, 19.7, p = 0.001), and the arousal index was reduced (2.5/h, IQR 2.3, 3.0 vs. 22.3/h, IQR 13.8, 30.0, p = 0.002), although total sleep time was similar between groups (621 min, IQR 464, 651 vs. 542.5 min, IQR 499, 582, p = 0.51). Remarkably, all six patients had MSLT latencies ≤8 min, indicating severe sleepiness. No parasomnia or sleep-disordered breathing was detected.
CONCLUSION
CONCLUSIONS
Central hypersomnia is a relevant characteristic of anti-GAD65-associated neurological syndromes.
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Informations de copyright
© 2023 The Authors. European Journal of Neurology published by John Wiley & Sons Ltd on behalf of European Academy of Neurology.
Références
Graus F, Titulaer MJ, Balu R, et al. A clinical approach to diagnosis of autoimmune encephalitis. Lancet Neurol. 2016;15(4):391-404. doi:10.1016/S1474-4422(15)00401-9
Devine MF, St Louis EK. Sleep disturbances associated with neurological autoimmunity. Neurotherapeutics. 2021;18(1):181-201. doi:10.1007/s13311-021-01020-x
Iranzo A. Sleep and neurological autoimmune diseases. Neuropsychopharmacology. 2020;45(1):129-140. doi:10.1038/s41386-019-0463-z
Muñoz-Lopetegi A, Graus F, Dalmau J, Santamaria J. Sleep disorders in autoimmune encephalitis. Lancet Neurol. 2020;19(12):1010-1022. doi:10.1016/S1474-4422(20)30341-0
Saiz A, Blanco Y, Sabater L, et al. Spectrum of neurological syndromes associated with glutamic acid decarboxylase antibodies: diagnostic clues for this association. Brain. 2008;131(10):2553-2563. doi:10.1093/brain/awn183
Vernet C, Arnulf I. Idiopathic hypersomnia with and without long sleep time: a controlled series of 75 patients. Sleep. 2009;32(6):753-759. doi:10.1093/sleep/32.6.753
American Academy of Sleep Medicine. The AASM Manual for the Scoring of Sleep and Associated Events: Rules, Terminology and Technical Specifications. American Academy of Sleep Medicine. 2020.
American Academy of Sleep Medicine. International Classification of Sleep Disorders. 3rd ed. American Academy of Sleep Medicine; 2014.
Mignot E, Lin X, Hesla PE, Dement WC, Guilleminault C, Grumet FC. A novel HLA DR17,DQ1, (DQA1-0102/DQB1-0602 positive), haplotype predisposing to narcolepsy in Caucasians. Sleep. 1993;16(8):764-765. doi:10.1093/sleep/16.8.764
Saper CB, Scammell TE, Lu J. Hypothalamic regulation of sleep and circadian rhythms. Nature. 2005;437(7063):1257-1263. doi:10.1038/nature04284
Liguori C, Toledo M, Kothare S. Effects of anti-seizure medications on sleep architecture and daytime sleepiness in patients with epilepsy: a literature review. Sleep Med Rev. 2021;60:101559. doi:10.1016/j.smrv.2021.101559
Ariño H, Muñoz-Lopetegi A, Martinez-Hernandez E, et al. Sleep disorders in anti-NMDAR encephalitis. Neurology. 2020;95(6):e671-e684. doi:10.1212/WNL.0000000000009987
Dalmau J. Clinical analysis of anti-MA2-associated encephalitis. Brain. 2004;127(8):1831-1844. doi:10.1093/brain/awh203
Dauvilliers Y, Bauer J, Rigau V, et al. Hypothalamic immunopathology in anti-MA-associated diencephalitis with narcolepsy-cataplexy. JAMA Neurol. 2013;12:1305-1310. doi:10.1001/jamaneurol.2013.2831
Brunet de Courssou JB, Testard P, Sallansonnet-Froment M, et al. Narcolepsy secondary to anti-MA2 encephalitis: two case reports. J Clin Sleep Med. 2023;19:837-841. doi:10.5664/jcsm.10448