Type 1 narcolepsy in anti-Hu antibodies mediated encephalitis: a case report

Vitiello, Maria; Antelmi, Elena; Pizza, Fabio; Postiglione, Emanuela; Poggi, Rosalba; Liguori, Rocco; Plazzi, Giuseppe

doi:10.1016/j.sleep.2018.02.003

Cited by 9 publications

(7 citation statements)

References 5 publications

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“…As discussed above, there have been several cases of low or absent CSF hypocretin-1 levels, some with fluctuation corresponding with symptom resolution or exacerbation. In most of the cases of low CSF hypocretin-1 level with neural-specific antibody positivity, the HLA haplotypes associated with idiopathic narcolepsy were absent [74,75,100,121,122].…”

Section: Sleep Work-upmentioning

confidence: 97%

“…Additional workup revealed ANNA-1 seropositivity and an indeterminate lung mass. Symptomatic therapy was ineffective [100].…”

Section: Anti-neuronal Nuclear Antibody Type-1 (Anna-1 Ie Hu) Syndromementioning

confidence: 99%

“…An autoimmune etiology should be considered when there are autoimmune risk factors or atypical, concurrent sleep manifestations, such as narcolepsy symptoms at an advanced age [100]. Sleep disorders are common in neurodegenerative conditions such as Alzheimer's disease (AD), dementia with Lewy bodies (DLB), Parkinson's disease, multiple system atrophy (MSA), and amyotrophic lateral sclerosis (ALS).…”

Section: Differential Diagnosismentioning

confidence: 99%

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Sleep Disturbances Associated with Neurological Autoimmunity

Devine

Louis

2021

Neurotherapeutics

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Associations between sleep disorders and neurological autoimmunity have been notably expanding recently. Potential immune-mediated etiopathogenesis has been proposed for various sleep disorders including narcolepsy, Kleine-Levin syndrome, and Morvan syndrome. Sleep manifestations are also common in various autoimmune neurological syndromes, but may be underestimated as overriding presenting (and potentially dangerous) neurological symptoms often require more urgent attention. Even so, sleep dysfunction has been described with various neural-specific antibody biomarkers, including IgLON5; leucine-rich, glioma-inactivated protein 1 (LGI1); contactin-associated protein 2 (CASPR2); N-methyl-D-aspartate (NMDA)-receptor; Ma2; dipeptidyl-peptidase-like protein-6 (DPPX); alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor (AMPA-R); anti-neuronal nuclear antibody type-1 (ANNA-1, i.e., Hu); anti-neuronal nuclear antibody type-2 (ANNA-2, i.e., Ri); gamma-aminobutyric acid (GABA)-B-receptor (GABA-B-R); metabotropic glutamate receptor 5 (mGluR5); and aquaporin-4 (AQP-4). Given potentially distinctive findings, it is possible that sleep testing could potentially provide objective biomarkers (polysomnography, quantitative muscle activity during REM sleep, cerebrospinal fluid hypocretin-1) to support an autoimmune diagnosis, monitor therapeutic response, or disease progression/relapse. However, more comprehensive characterization of sleep manifestations is needed to better understand the underlying sleep disruption with neurological autoimmunity.

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Section: Sleep Work-upmentioning

confidence: 97%

“…Additional workup revealed ANNA-1 seropositivity and an indeterminate lung mass. Symptomatic therapy was ineffective [100].…”

Section: Anti-neuronal Nuclear Antibody Type-1 (Anna-1 Ie Hu) Syndromementioning

confidence: 99%

Section: Differential Diagnosismentioning

confidence: 99%

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Sleep Disturbances Associated with Neurological Autoimmunity

Devine

Louis

2021

Neurotherapeutics

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“…multiple sclerosis) or tumoural lesions of the hypothalamus associated with EDS, and can have a favourable course along with resolution of the primary brain damage, as well as with antibody‐mediated disorders such as neuromyelitis optica with anti‐aquaporin‐4 antibodies or limbic encephalitis of undetermined origin responsive to immunological approaches (Kanbayashi et al, 2011; Nishino & Kanbayashi, 2005). Otherwise, secondary narcolepsy can be paraneoplastic and tell‐tale the presence of a tumour located outside of the central nervous system (Dauvilliers et al, 2013; Landolfi & Nadkarni, 2003; Overeem et al, 2004; Vitiello et al, 2018), with the link between the primary neoplasm (e.g. testicular, pulmonal) and the onset of narcolepsy being the presence of specific antibodies such as anti‐Ma2 or anti‐Hu (Dauvilliers et al, 2013; Landolfi & Nadkarni, 2003; Overeem et al, 2004; Vitiello et al, 2018) or unknown (Rossi et al, 2021).…”

Section: Sleep and Sleep Disturbancesmentioning

confidence: 99%

The orexin story, sleep and sleep disturbances

Pizza

Barateau

Dauvilliers

et al. 2022

Journal of Sleep Research

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Summary The orexins, also known as hypocretins, are two neuropeptides (orexin A and B or hypocretin 1 and 2) produced by a few thousand neurons located in the lateral hypothalamus that were independently discovered by two research groups in 1998. Those two peptides bind two receptors (orexin/hypocretin receptor 1 and receptor 2) that are widely distributed in the brain and involved in the central physiological regulation of sleep and wakefulness, orexin receptor 2 having the major role in the maintenance of arousal. They are also implicated in a multiplicity of other functions, such as reward seeking, energy balance, autonomic regulation and emotional behaviours. The destruction of orexin neurons is responsible for the sleep disorder narcolepsy with cataplexy (type 1) in humans, and a defect of orexin signalling also causes a narcoleptic phenotype in several animal species. Orexin discovery is unprecedented in the history of sleep research, and pharmacological manipulations of orexin may have multiple therapeutic applications. Several orexin receptor antagonists were recently developed as new drugs for insomnia, and orexin agonists may be the next‐generation drugs for narcolepsy. Given the broad range of functions of the orexin system, these drugs might also be beneficial for treating various conditions other than sleep disorders in the near future.

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“…HLA DQB1*0602 allele positivity is also often detected in type 1 patients, suggesting that the disease has a certain genetic background. The symptoms of type 2 narcolepsy are similar to those of type 1, but without cataplexy, and the level of hypocretin in the CSF is normal (6). Its pathogenesis is unknown, but it may be related to the partial loss of hypocretin neurons.…”

Section: Introductionmentioning

confidence: 98%

Type 1 narcolepsy without cataplexy and treatment progress: a case report

Huang

Ding

et al. 2021

Ann Palliat Med

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Recent studies have suggested that REM-related OSA is also closely related to narcolepsy (4).According to the International Classification of Sleep Disorders III, narcolepsy is classified as Types 1 and 2. Type 1 narcolepsy is mainly characterized by excessive sleepiness and cataplexy caused by emotional factors (i.e., laughter and anger), and may also manifest as sleep paralysis, hallucinations before falling asleep, nighttime sleep disturbance,

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Type 1 narcolepsy in anti-Hu antibodies mediated encephalitis: a case report

Cited by 9 publications

References 5 publications

Sleep Disturbances Associated with Neurological Autoimmunity

Sleep Disturbances Associated with Neurological Autoimmunity

The orexin story, sleep and sleep disturbances

Type 1 narcolepsy without cataplexy and treatment progress: a case report

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