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Sleep disorders and polysomnography findings in patients with autoimmune encephalitis

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Abstract

Background

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).

Methods

Seventeen patients with diagnosed AE and acute or subacute onset sleep complaints who underwent video-electroencephalography-PSG recordings in our tertiary center were investigated.

Results

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.

Conclusions

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.

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Abbreviations

AASM:

American Academy of Sleep Medicine

AE:

Autoimmune encephalitis

AHI:

Apnea–hypopnea index

ANNA-2:

Anti-neuronal nuclear antibody 2

CASPR2:

Contactin-associated protein-2

CRMP-5:

Collapsin-response mediator protein 5

CSA:

Central sleep apnea

CSB:

Cheyne–Stokes breathing

CSF:

Cerebrospinal fluid

DNER:

Delta/notch-like epidermal growth factor-related receptor

DPPX:

Dipeptidyl-peptidase-like protein- 6

EDS:

Excessive daytime sleepiness

EEG:

Electroencephalography

GAD 65:

Glutamic acid decarboxylase 65

IQR:

Interquartile range

IVIG:

Intravenous immunoglobulin

LGI1:

Leucine-rich glioma-inactivated 1

MRI:

Magnetic resonance imaging

NMDAR:

N-methyl-d-aspartate receptor

NREM:

Non-rapid eye movement

OSA:

Obstructive sleep apnea

PCA-1:

Purkinje cell antibody 1

PLM:

Periodic limb movements

PSG:

Polysomnography

REM:

Rapid eye movement

SE:

Sleep efficiency

SOX-1:

Sry-like high mobility group box 1

SPSS:

Statistical Package for the Social Sciences

SRBD:

Sleep-related breathing disorder

TST:

Total sleep time

VGKC:

Voltage-gated potassium channel complex

Zic4:

Zinc finger protein 4

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Authors and Affiliations

Authors

Contributions

All authors contributed to the study conception and design. Material preparation, data collection, and analysis were performed by IE and FIT. The first draft of the manuscript was written by IE and FIT, and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

Conceptualization, IE and FIT; methodology, IE and FIT; formal analysis and investigation, IE and FIT; writing — original draft preparation, IE and FIT; writing — review and editing, IE, BE, ES, SS, and FIT; funding acquisition, none; resources, IE, BE, ES, SS, and FIT; and supervision, BE, ES, SS, and FIT.

Corresponding author

Correspondence to Irem Erkent.

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Ethics approval

This study was performed in line with the principles of the Declaration of Helsinki. Approval was granted by the Ethics Committee of Hacettepe University Non-interventional Clinical Researches Ethics Board.

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Written informed consent was obtained from all participants.

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The authors declare no competing interests.

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Erkent, I., Elibol, B., Saka, E. et al. Sleep disorders and polysomnography findings in patients with autoimmune encephalitis. Neurol Sci 44, 1351–1360 (2023). https://doi.org/10.1007/s10072-022-06513-x

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