Post-H1N1 Narcolepsy-Cataplexy

Dauvilliers, Yves; Montplaisir, Jacques; Cochen, Valéerie; Désautels, Alex; Einen, Mali; Li, Gang; Kawashima, M.; Bayard, Sophie; Monaca, Christelle; Tiberge, M.; Filipini, Daniel; Tripathy, Asit K.; Nguyễn, Bich Hồng; Kotagal, Suresh; Mignot, Emmanuel

doi:10.1093/sleep/33.11.1428

Cited by 197 publications

(142 citation statements)

References 17 publications

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“…Given that selection bias is likely to have led to the investigator allocating more severely affected children for inclusion in the IVIg group, it is tempting to speculate that these children might have fared worse had they not received IVIg. Of note, those treated with IVIg had a much higher exposure to H1N1 vaccination (15 of 22) and a potentially different pathogenesis and disease course 39 compared with the control group (2 of 30), which largely comprised patients with sporadic onset. Differing vaccine coadjuvants may modulate immunological response, as illustrated by cross-reactivity between H1N1 nucleoprotein serum antibodies and the hypocretin receptor 2 following Pandremix, but not Focetria vaccination.…”

mentioning

confidence: 99%

Intravenous Immunoglobulin Therapy in Pediatric Narcolepsy: A Nonrandomized, Open-Label, Controlled, Longitudinal Observational Study

Lecendreux

Berthier

Corny

et al. 2017

Journal of Clinical Sleep Medicine

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Study Objectives: Previous case reports of intravenous immunoglobulins (IVIg) in pediatric narcolepsy have shown contradictory results. Methods: This was a nonrandomized, open-label, controlled, longitudinal observational study of IVIg use in pediatric narcolepsy with retrospective data collection from medical files obtained from a single pediatric national reference center for the treatment of narcolepsy in France. Of 56 consecutively referred patients with narcolepsy, 24 received IVIg (3 infusions administered at 1-mo intervals) in addition to standard care (psychostimulants and/or anticataplectic agents), and 32 continued on standard care alone (controls). Results: For two patients in each group, medical files were unavailable. Of the 22 IVIg patients, all had cerebrospinal fluid (CSF) hypocretin ≤ 110 pg/mL and were HLA-DQB1*06:02 positive. Of the 30 control patients, 29 were HLA-DQB1*06:02 positive and of those with available CSF measurements, all 12 had hypocretin ≤ 110 pg/mL. Compared with control patients, IVIg patients had shorter disease duration, shorter latency to sleep onset, and more had received H1N1 vaccination. Mean (standard deviation) follow-up length was 2.4 (1.1) y in the IVIg group and 3.9 (1.7) y in controls. In multivariate-adjusted linear mixed-effects analyses of change from baseline in Ullanlinna Narcolepsy Scale (UNS) scores, high baseline UNS, but not IVIg treatment, was associated with a reduction in narcolepsy symptoms. On time-to-event analysis, among patients with high baseline UNS scores, control patients achieved a UNS score < 14 (indicating remission) less rapidly than IVIg patients (adjusted hazard ratio 0.18; 95% confidence interval: 95% confidence interval: 0.03, 0.95; p = 0.043). Shorter or longer disease duration did not influence treatment response in any analysis. Conclusions: Overall, narcolepsy symptoms were not significantly reduced by IVIg. However, in patients with high baseline symptoms, a subset of IVIgtreated patients achieved remission more rapidly than control patients. I NTRO DUCTI O NNarcolepsy is a debilitating and currently an incurable neurological condition with a characteristic constellation of symptoms, including excessive daytime sleepiness (EDS), cataplexy, sleep paralysis, hypnagogic hallucinations, and disturbed nocturnal sleep.Until recently, and at the time patients in this study were treated, the disorder was categorized into narcolepsy with cataplexy and narcolepsy without cataplexy according to the revised second edition of the International Classification of Sleep Disorders (ICSD).1 ICSD-2-revised has now been replaced by ICSD-3, which classifies narcolepsy into type 1 and type 2 narcolepsy. 2The majority of patients with type 1 narcolepsy have hypocretin deficiency (cerebrospinal fluid [CSF] hypocretin-1 level ≤ 110 pg/ mL), which is considered to result from an autoimmune-mediated loss of hypocretin-secreting neurons in those who are genetically predisposed (most commonly those with human leucocyte antigen [HLA] DQB1*06:02 haplotype). The...

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Intravenous Immunoglobulin Therapy in Pediatric Narcolepsy: A Nonrandomized, Open-Label, Controlled, Longitudinal Observational Study

Lecendreux

Berthier

Corny

et al. 2017

Journal of Clinical Sleep Medicine

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“…‡ SUMMARY Narcolepsy with cataplexy: an autoimmune disease? Narcolepsy type 1 (also named narcolepsy-cataplexy or hypocretin deficiency syndrome) is a rare sleep disorder dans d'autres pays utilisant également le vaccin, comme l'Irlande, le Royaume-Uni et la France [42,[44][45][46]. Curieusement, une telle augmentation de l'incidence de la narcolepsie n'a pas été observée en Amérique du Nord ou dans certains pays d'Amérique du Sud où Arepanrix (GSK, Canada), un vaccin très similaire à Pandemrix, a été utilisé [47][48][49] …”

Section: Resultsunclassified

La narcolepsie avec cataplexie : une maladie auto-immune ?

Jacob

Dauvilliers

2014

Med Sci (Paris)

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“…Notably, a squalene-adjuvanted pandemic influenza vaccine (Pandemirix Ò , GSK) was recently found to be associated with an increased risk of narcolepsy in young children, highlighting the uncertain safety of oil-emulsion adjuvants in less studied patient populations such as young children and pregnant women. 73,74 As extensive safety data is available on use of unadjuvanted seasonal influenza vaccines in pregnancy, it is logical to continue their use in pregnant women until such time as an alternative adjuvanted seasonal influenza vaccine is shown to be more effective but at least as safe in pregnant women.…”

Section: Safety Aspects Of Maternal Influenza Immunizationmentioning

confidence: 99%