Long-term follow-up on the effects of sodium oxybate on daytime sleepiness and sleep architecture in patients with narcolepsy type 1.

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Abstract

Sodium oxybate (SXB) was administered for the first time in 1979 in 16 patients with narcolepsy with cataplexy (NT1) that improved up to 20 months.

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Most cited references 17

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European guideline and expert statements on the management of narcolepsy in adults and children.

Claudio Bassetti, Ulf Kallweit, Luca Vignatelli … (2021)

Narcolepsy is an uncommon hypothalamic disorder of presumed autoimmune origin that usually requires lifelong treatment. This paper aims to provide evidence-based guidelines for the management of narcolepsy in both adults and children.

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A missense mutation in myelin oligodendrocyte glycoprotein as a cause of familial narcolepsy with cataplexy.

Hyun Hor, Luca Bartesaghi, Zoltan Kutalik … (2011)

Narcolepsy is a rare sleep disorder characterized by excessive daytime sleepiness and cataplexy. Familial narcolepsy accounts for less than 10% of all narcolepsy cases. However, documented multiplex families are very rare and causative mutations have not been identified to date. To identify a causative mutation in familial narcolepsy, we performed linkage analysis in the largest ever reported family, which has 12 affected members, and sequenced coding regions of the genome (exome sequencing) of three affected members with narcolepsy and cataplexy. We successfully mapped a candidate locus on chromosomal region 6p22.1 (LOD score ¼ 3.85) by linkage analysis. Exome sequencing identified a missense mutation in the second exon of MOG within the linkage region. A c.398C>G mutation was present in all affected family members but absent in unaffected members and 775 unrelated control subjects. Transient expression of mutant myelin oligodendrocyte glycoprotein (MOG) in mouse oligodendrocytes showed abnormal subcellular localization, suggesting an altered function of the mutant MOG. MOG has recently been linked to various neuropsychiatric disorders and is considered as a key autoantigen in multiple sclerosis and in its animal model, experimental autoimmune encephalitis. Our finding of a pathogenic MOG mutation highlights a major role for myelin and oligodendrocytes in narcolepsy and further emphasizes glial involvement in neurodegeneration and neurobehavioral disorders. [corrected]. Copyright © 2011 The American Society of Human Genetics. Published by Elsevier Inc. All rights reserved.

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A pilot study on the effects of sodium oxybate on sleep architecture and daytime alertness in narcolepsy.

R Kovacević-Ristanović, Jacques Montplaisir, Kevin J. Black … (2004)

To measure the effect of nocturnal sodium oxybate administration on sleep architecture in patients with narcolepsy. Open-label study. Four accredited sleep clinics. 25 adult patients with narcolepsy-cataplexy. Patients were weaned from previously used anticataplectic medications and administered increasing nightly doses of sodium oxybate over a 10-week period: 4.5 g for 4 weeks, 6 g for 2 weeks, 7.5 g for 2 weeks, and 9 g for 2 weeks. The effect of sodium oxybate was measured using nocturnal polysomnograms, the Epworth Sleepiness Scale, the Maintenance of Wakefulness Test, and a narcolepsy symptoms questionnaire. The nightly administration of sodium oxybate produced dose-related increases in slow-wave sleep and delta power, rapid eye movement sleep increased initially and then decreased in a dose-related manner, nocturnal awakenings decreased, and daytime sleep latency increased. Significant improvements in daytime symptoms were measured by the Maintenance of Wakefulness Test, the Epworth Sleepiness Scale, and the narcolepsy symptom questionnaire. Nocturnal administration of sodium oxybate in patients with narcolepsy produces significant improvements in sleep architecture, which coincide with significant improvements in daytime functioning.

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Author and article information

Journal

Journal ID (iso-abbrev): Rev Neurol

Title: Revista de neurologia

Publisher: Viguera Editores SLU

ISSN (Electronic): 1576-6578

ISSN (Print): 0210-0010

Publication date (Electronic): Jan 16 2023

Volume: 76

Issue: 2

Affiliations

[1 ] Hospital General Universitario Gregorio Marañón, Madrid, España.

[2 ] Instituto de Investigación Gregorio Marañón. Universidad Complutense de Madrid, Madrid, España.

[3 ] Hospital Ruber Internacional, Madrid, España.

[4 ] Hospital Universitario Niño Jesús, Madrid, España.

[5 ] Universidad San Pablo-CEU, Madrid, España.

Article

Publisher Item ID: rn2022315

DOI: 10.33588/rn.7602.2022315

PubMed ID: 36631962

SO-VID: 1f3f6af0-4c31-4d56-b218-3a98d1d8c96f

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Long-term follow-up on the effects of sodium oxybate on daytime sleepiness and sleep architecture in patients with narcolepsy type 1.

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Abstract

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Higher order chromatin architecture

Most cited references 17

European guideline and expert statements on the management of narcolepsy in adults and children.

A missense mutation in myelin oligodendrocyte glycoprotein as a cause of familial narcolepsy with cataplexy.

A pilot study on the effects of sodium oxybate on sleep architecture and daytime alertness in narcolepsy.

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