Polysomnographic and actigraphic characteristics of patients with H1N1-vaccine-related and sporadic narcolepsy

Alakuijala, Anniina; Sarkanen, Tomi; Partinen, Markku

doi:10.1016/j.sleep.2014.07.024

Cited by 20 publications

(13 citation statements)

References 41 publications

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“…On the other hand, a comparison between Finnish children with pNT1 and Italian children with NT1 appears to show similar clinical pictures, although subjects with pNT1 often had increased nocturnal sleep disturbance [14]. In a recent study on polysomnographic and actigraphic characteristics of pNT1 and NT1, we found that NT1 subjects had delayed sleep-wake rhythms compared to pNT1, but otherwise we did not see dramatic differences [15].…”

Section: Introductionmentioning

confidence: 47%

Clinical course of H1N1-vaccine-related narcolepsy

2016

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Objective: To follow and analyze the clinical course and quality of life of Pandemrix H1N1-vaccinerelated narcolepsy (pNT1). Methods: Twenty-six drug-naïve confirmed pNT1 subjects completed Epworth Sleepiness Scale (ESS), Ullanlinna Narcolepsy Scale (UNS), Swiss Narcolepsy Scale (SNS), Rimon's Brief Depression scale (RDS), and WHO-5 Well-being index questionnaires near the disease onset and in a follow-up a minimum of two years later. The number of cataplexies and body mass index (BMI) were recorded. The effects of hypocretin-1 levels and sleep recording results were analyzed. The findings at the follow-up visit were compared with 25 non-vaccine-related type 1 narcolepsy (NT1) subjects. Results: In pNT1, RDS score decreased significantly (mean 10.2, SD 4.7 vs mean 6.7, SD 4.5, p = 0.003). Median of BMI increased from 20.8 kg m −2 to 23.4 kg m −2 , p < 0.001. There were no significant differences in other sleep scores. However, deviation and range in questionnaire scores at the follow-up were wide. Subjects with very low or undetectable hypocretin-1 levels had worse scores in UNS (mean 26.4, SD 6.95 vs mean 19.1, SD 3.83, p = 0.006) and ESS (mean 17.9, SD = 4.29 vs mean 14.1, SD = 3.70, p = 0.047) than those with hypocretin-1 levels of 20-110 pg/mL. Most disabling symptoms were excessive daytime sleepiness and disturbed sleep. There were no significant differences between the scores in pNT1 and NT1. Conclusions: Clinical course of pNT1 is heterogeneous but the evolution of pNT1 seems similar to NT1. Lower hypocretin levels in pNT1 are associated with a more severe phenotype.

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Section: Introductionmentioning

confidence: 47%

Clinical course of H1N1-vaccine-related narcolepsy

2016

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“…Recently, several studies have successfully applied actigraphy in the clinical work-up of adult and pediatric NT1 [17][18][19].…”

Section: Introductionmentioning

confidence: 99%

In-field assessment of sodium oxybate effect in pediatric type 1 narcolepsy: an actigraphic study

Filardi

Pizza

Antelmi

et al. 2018

Sleep

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Actigraphy objectively documented changes in nocturnal sleep quality and diurnal napping behavior after 1 year of SXB treatment, thus representing a valid approach to ecologically assess SXB treatment effect on NT1 children's sleep/wake profile. NT1 symptoms severity and children's anthropometric features also changed as expected. Actigraphy offers the possibility to longitudinally follow up children and has potential to become a key tool to tailor treatment in pediatric patients.

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“…The affinity analysis was restricted to childhood patients not only primarily because they had higher levels of antibodies against HA2009 (27), NP-CA2009, and NP-PR1934 but also because childhood age at onset was unique to the Pandemrixassociated onset of narcolepsy (4,37,39,41,45). It is noteworthy that the association between antibody levels and age was only positive for NP-PR1934 among the controls.…”

Section: Discussionmentioning

confidence: 99%

Antibody Affinity Against 2009 A/H1N1 Influenza and Pandemrix Vaccine Nucleoproteins Differs Between Childhood Narcolepsy Patients and Controls

et al. 2017

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Increased narcolepsy incidence was observed in Sweden following the 2009 influenza vaccination with Pandemrix. A substitution of the 2009 nucleoprotein for the 1934 variant has been implicated in narcolepsy development. The aims were to determine (a) antibody levels toward wild-type A/H1N1-2009[A/California/04/2009(H1N1)] (NP-CA2009) and Pandemrix-[A/Puerto Rico/8/1934(H1N1)] (NP-PR1934) nucleoproteins in 43 patients and 64 age-matched controls; (b) antibody affinity in reciprocal competitive assays in 11 childhood narcolepsy patients compared with 21 age-matched controls; and (c) antibody levels toward wild-type A/H1N1-2009[A/California/04/2009(H1N1)] (H1N1 NS1), not a component of the Pandemrix vaccine. In vitro transcribed and translated S-methionine-labeled H1N1 influenza A virus proteins were used in radiobinding reciprocal competition assays to estimate antibody levels and affinity (Kd). Childhood patients had higher NP-CA2009 (p = 0.0339) and NP-PR1934 (p = 0.0246) antibody levels compared with age-matched controls. These childhood controls had lower NP-CA2009 (p = 0.0221) and NP-PR1934 (p = 0.00619) antibodies compared with controls 13 years or older. In contrast, in patients 13 years or older, the levels of NP-PR1934 (p = 0.279) and NP-CA2009 (p = 0.0644) antibodies did not differ from the older controls. Childhood antibody affinity (Kd) against NP-CA2009 was comparable between controls (68 ng/mL) and patients (74 ng/mL; p = 0.21) with NP-CA2009 and NP-PR1934 displacement (controls: 165 ng/mL; patients: 199 ng/mL; p = 0.48). In contrast, antibody affinity against NP-PR1934 was higher in controls with either NP-PR1934 (controls: 9 ng/mL; patients: 20 ng/mL; p = 0.0031) or NP-CA2009 (controls: 14 ng/mL; patients: 23 ng/mL; p = 0.0048). A/H1N1-NS1 antibodies were detected in 0/43 of the narcolepsy patients compared with 3/64 (4.7%) controls (p = 0.272). Similarly, none (0/11) of the childhood patients and 1/21 (4.8%) of the childhood controls had A/H1N1-NS1 antibodies. The higher antibody affinities against NP-PR1934 in controls suggest better protection against wild-type virus. In contrast, the reduced NP-PR1934 antibody affinities among childhood narcolepsy patients suggest poor protection from the wild-type A/H1N1 virus and possibly increased risk for viral damage.

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Polysomnographic and actigraphic characteristics of patients with H1N1-vaccine-related and sporadic narcolepsy

Cited by 20 publications

References 41 publications

Clinical course of H1N1-vaccine-related narcolepsy

Clinical course of H1N1-vaccine-related narcolepsy

In-field assessment of sodium oxybate effect in pediatric type 1 narcolepsy: an actigraphic study

Antibody Affinity Against 2009 A/H1N1 Influenza and Pandemrix Vaccine Nucleoproteins Differs Between Childhood Narcolepsy Patients and Controls

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