New 2013 incidence peak in childhood narcolepsy: more than vaccination?

Zhongxing Zhang; Jari Gool; Rolf Fronczek; Yves Dauvilliers; Claudio L A Bassetti; Geert Mayer; Giuseppe Plazzi; Fabio Pizza; Joan Santamaria; Markku Partinen; Sebastiaan Overeem; Rosa Peraita-Adrados; Antonio Martins da Silva; Karel Sonka; Rafael Del Rio-Villegas; Raphael Heinzer; Aleksandra Wierzbicka; Peter Young; Birgit Högl; Mauro Manconi; Eva Feketeova; Johannes Mathis; Teresa Paiva; Francesca Canellas; Michel Lecendreux; Christian R Baumann; Gert Jan Lammers; Ramin Khatami

doi:10.1093/sleep/zsaa172

New 2013 incidence peak in childhood narcolepsy: more than vaccination?

Zhongxing Zhang, Jari Gool, Rolf Fronczek, Yves Dauvilliers, Claudio L A Bassetti, Geert Mayer, Giuseppe Plazzi, Fabio Pizza, Joan Santamaria, Markku Partinen, Sebastiaan Overeem, Rosa Peraita-Adrados, Antonio Martins da Silva, Karel Sonka, Rafael Del Rio-Villegas, Raphael Heinzer, Aleksandra Wierzbicka, Peter Young, Birgit Högl, Mauro ManconiEva Feketeova, Johannes Mathis, Teresa Paiva, Francesca Canellas, Michel Lecendreux, Christian R Baumann, Gert Jan Lammers, Ramin Khatami

Istituto Scienze Neurologiche

Research output: Contribution to journal › Article › peer-review

Abstract

Increased incidence rates of narcolepsy type-1 (NT1) have been reported world-wide after the 2009-2010 H1N1 influenza pandemic (pH1N1). While some European countries found an association between the NT1 incidence increase and the H1N1 vaccination Pandemrix, reports from Asian countries suggested the H1N1 virus itself to be linked to the increased NT1 incidence. Using robust data-driven modelling approaches, i.e., locally estimated scatterplot smoothing methods, we analyzed the number of de-novo NT1 cases (n= 508) in the last two decades using the European Narcolepsy Network database. We confirmed the peak of NT1 incidence in 2010, i.e., 2.54-fold (95% confidence interval [CI]: [2.11, 3.19]) increase in NT1 onset following 2009-2010 pH1N1. This peak in 2010 was found in both childhood NT1 (2.75-fold increase, 95% CI: [1.95, 4.69]) and adulthood NT1 (2.43-fold increase, 95% CI: [2.05, 2.97]). In addition, we identified a new peak in 2013 that is age-specific for children/adolescents (i.e., 2.09-fold increase, 95% CI: [1.52, 3.32]). Most of these children/adolescents were HLA DQB1*06:02 positive and showed a subacute disease onset consistent with an immune-mediated type of narcolepsy. The new 2013 incidence peak is likely not related to Pandemrix as it was not used after 2010. Our results suggest that the increased NT1 incidence after 2009-2010 pH1N1 is not unique and our study provides an opportunity to develop new hypotheses, e.g., considering other (influenza) viruses or epidemiological events to further investigate the pathophysiology of immune-mediated narcolepsy.

Original language	English
Journal	Sleep
DOIs	https://doi.org/10.1093/sleep/zsaa172
Publication status	E-pub ahead of print - Sep 10 2020

Access to Document

10.1093/sleep/zsaa172

Cite this

Zhang, Z., Gool, J., Fronczek, R., Dauvilliers, Y., Bassetti, C. L. A., Mayer, G., Plazzi, G., Pizza, F., Santamaria, J., Partinen, M., Overeem, S., Peraita-Adrados, R., da Silva, A. M., Sonka, K., Del Rio-Villegas, R., Heinzer, R., Wierzbicka, A., Young, P., Högl, B., ... Khatami, R. (2020). New 2013 incidence peak in childhood narcolepsy: more than vaccination? Sleep. https://doi.org/10.1093/sleep/zsaa172

New 2013 incidence peak in childhood narcolepsy : more than vaccination? / Zhang, Zhongxing; Gool, Jari; Fronczek, Rolf; Dauvilliers, Yves; Bassetti, Claudio L A; Mayer, Geert; Plazzi, Giuseppe ; Pizza, Fabio; Santamaria, Joan; Partinen, Markku; Overeem, Sebastiaan; Peraita-Adrados, Rosa; da Silva, Antonio Martins; Sonka, Karel; Del Rio-Villegas, Rafael; Heinzer, Raphael; Wierzbicka, Aleksandra; Young, Peter; Högl, Birgit; Manconi, Mauro; Feketeova, Eva; Mathis, Johannes; Paiva, Teresa; Canellas, Francesca; Lecendreux, Michel; Baumann, Christian R; Lammers, Gert Jan; Khatami, Ramin.

In: Sleep, 10.09.2020.

Research output: Contribution to journal › Article › peer-review

Zhang, Z, Gool, J, Fronczek, R, Dauvilliers, Y, Bassetti, CLA, Mayer, G, Plazzi, G , Pizza, F, Santamaria, J, Partinen, M, Overeem, S, Peraita-Adrados, R, da Silva, AM, Sonka, K, Del Rio-Villegas, R, Heinzer, R, Wierzbicka, A, Young, P, Högl, B, Manconi, M, Feketeova, E, Mathis, J, Paiva, T, Canellas, F, Lecendreux, M, Baumann, CR, Lammers, GJ & Khatami, R 2020, 'New 2013 incidence peak in childhood narcolepsy: more than vaccination?', Sleep. https://doi.org/10.1093/sleep/zsaa172

Zhang, Zhongxing ; Gool, Jari ; Fronczek, Rolf ; Dauvilliers, Yves ; Bassetti, Claudio L A ; Mayer, Geert ; Plazzi, Giuseppe ; Pizza, Fabio ; Santamaria, Joan ; Partinen, Markku ; Overeem, Sebastiaan ; Peraita-Adrados, Rosa ; da Silva, Antonio Martins ; Sonka, Karel ; Del Rio-Villegas, Rafael ; Heinzer, Raphael ; Wierzbicka, Aleksandra ; Young, Peter ; Högl, Birgit ; Manconi, Mauro ; Feketeova, Eva ; Mathis, Johannes ; Paiva, Teresa ; Canellas, Francesca ; Lecendreux, Michel ; Baumann, Christian R ; Lammers, Gert Jan ; Khatami, Ramin. / New 2013 incidence peak in childhood narcolepsy : more than vaccination?. In: Sleep. 2020.

@article{78f60ab7761a4a98b06b0e2dd599a49d,

title = "New 2013 incidence peak in childhood narcolepsy: more than vaccination?",

abstract = "Increased incidence rates of narcolepsy type-1 (NT1) have been reported world-wide after the 2009-2010 H1N1 influenza pandemic (pH1N1). While some European countries found an association between the NT1 incidence increase and the H1N1 vaccination Pandemrix, reports from Asian countries suggested the H1N1 virus itself to be linked to the increased NT1 incidence. Using robust data-driven modelling approaches, i.e., locally estimated scatterplot smoothing methods, we analyzed the number of de-novo NT1 cases (n= 508) in the last two decades using the European Narcolepsy Network database. We confirmed the peak of NT1 incidence in 2010, i.e., 2.54-fold (95% confidence interval [CI]: [2.11, 3.19]) increase in NT1 onset following 2009-2010 pH1N1. This peak in 2010 was found in both childhood NT1 (2.75-fold increase, 95% CI: [1.95, 4.69]) and adulthood NT1 (2.43-fold increase, 95% CI: [2.05, 2.97]). In addition, we identified a new peak in 2013 that is age-specific for children/adolescents (i.e., 2.09-fold increase, 95% CI: [1.52, 3.32]). Most of these children/adolescents were HLA DQB1*06:02 positive and showed a subacute disease onset consistent with an immune-mediated type of narcolepsy. The new 2013 incidence peak is likely not related to Pandemrix as it was not used after 2010. Our results suggest that the increased NT1 incidence after 2009-2010 pH1N1 is not unique and our study provides an opportunity to develop new hypotheses, e.g., considering other (influenza) viruses or epidemiological events to further investigate the pathophysiology of immune-mediated narcolepsy.",

author = "Zhongxing Zhang and Jari Gool and Rolf Fronczek and Yves Dauvilliers and Bassetti, {Claudio L A} and Geert Mayer and Giuseppe Plazzi and Fabio Pizza and Joan Santamaria and Markku Partinen and Sebastiaan Overeem and Rosa Peraita-Adrados and {da Silva}, {Antonio Martins} and Karel Sonka and {Del Rio-Villegas}, Rafael and Raphael Heinzer and Aleksandra Wierzbicka and Peter Young and Birgit H{\"o}gl and Mauro Manconi and Eva Feketeova and Johannes Mathis and Teresa Paiva and Francesca Canellas and Michel Lecendreux and Baumann, {Christian R} and Lammers, {Gert Jan} and Ramin Khatami",

note = "Ricercatore distaccato presso IRCCS a seguito Convenzione esclusiva con Universit{\`a} di Bologna (Plazzi Giuseppe, Pizza Fabio) ",

year = "2020",

month = sep,

day = "10",

doi = "10.1093/sleep/zsaa172",

language = "English",

journal = "Sleep",

issn = "0161-8105",

publisher = "American Academy of Sleep Medicine",

}

TY - JOUR

T1 - New 2013 incidence peak in childhood narcolepsy

T2 - more than vaccination?

AU - Zhang, Zhongxing

AU - Gool, Jari

AU - Fronczek, Rolf

AU - Dauvilliers, Yves

AU - Bassetti, Claudio L A

AU - Mayer, Geert

AU - Plazzi, Giuseppe

AU - Pizza, Fabio

AU - Santamaria, Joan

AU - Partinen, Markku

AU - Overeem, Sebastiaan

AU - Peraita-Adrados, Rosa

AU - da Silva, Antonio Martins

AU - Sonka, Karel

AU - Del Rio-Villegas, Rafael

AU - Heinzer, Raphael

AU - Wierzbicka, Aleksandra

AU - Young, Peter

AU - Högl, Birgit

AU - Manconi, Mauro

AU - Feketeova, Eva

AU - Mathis, Johannes

AU - Paiva, Teresa

AU - Canellas, Francesca

AU - Lecendreux, Michel

AU - Baumann, Christian R

AU - Lammers, Gert Jan

AU - Khatami, Ramin

N1 - Ricercatore distaccato presso IRCCS a seguito Convenzione esclusiva con Università di Bologna (Plazzi Giuseppe, Pizza Fabio)

PY - 2020/9/10

Y1 - 2020/9/10

N2 - Increased incidence rates of narcolepsy type-1 (NT1) have been reported world-wide after the 2009-2010 H1N1 influenza pandemic (pH1N1). While some European countries found an association between the NT1 incidence increase and the H1N1 vaccination Pandemrix, reports from Asian countries suggested the H1N1 virus itself to be linked to the increased NT1 incidence. Using robust data-driven modelling approaches, i.e., locally estimated scatterplot smoothing methods, we analyzed the number of de-novo NT1 cases (n= 508) in the last two decades using the European Narcolepsy Network database. We confirmed the peak of NT1 incidence in 2010, i.e., 2.54-fold (95% confidence interval [CI]: [2.11, 3.19]) increase in NT1 onset following 2009-2010 pH1N1. This peak in 2010 was found in both childhood NT1 (2.75-fold increase, 95% CI: [1.95, 4.69]) and adulthood NT1 (2.43-fold increase, 95% CI: [2.05, 2.97]). In addition, we identified a new peak in 2013 that is age-specific for children/adolescents (i.e., 2.09-fold increase, 95% CI: [1.52, 3.32]). Most of these children/adolescents were HLA DQB1*06:02 positive and showed a subacute disease onset consistent with an immune-mediated type of narcolepsy. The new 2013 incidence peak is likely not related to Pandemrix as it was not used after 2010. Our results suggest that the increased NT1 incidence after 2009-2010 pH1N1 is not unique and our study provides an opportunity to develop new hypotheses, e.g., considering other (influenza) viruses or epidemiological events to further investigate the pathophysiology of immune-mediated narcolepsy.

AB - Increased incidence rates of narcolepsy type-1 (NT1) have been reported world-wide after the 2009-2010 H1N1 influenza pandemic (pH1N1). While some European countries found an association between the NT1 incidence increase and the H1N1 vaccination Pandemrix, reports from Asian countries suggested the H1N1 virus itself to be linked to the increased NT1 incidence. Using robust data-driven modelling approaches, i.e., locally estimated scatterplot smoothing methods, we analyzed the number of de-novo NT1 cases (n= 508) in the last two decades using the European Narcolepsy Network database. We confirmed the peak of NT1 incidence in 2010, i.e., 2.54-fold (95% confidence interval [CI]: [2.11, 3.19]) increase in NT1 onset following 2009-2010 pH1N1. This peak in 2010 was found in both childhood NT1 (2.75-fold increase, 95% CI: [1.95, 4.69]) and adulthood NT1 (2.43-fold increase, 95% CI: [2.05, 2.97]). In addition, we identified a new peak in 2013 that is age-specific for children/adolescents (i.e., 2.09-fold increase, 95% CI: [1.52, 3.32]). Most of these children/adolescents were HLA DQB1*06:02 positive and showed a subacute disease onset consistent with an immune-mediated type of narcolepsy. The new 2013 incidence peak is likely not related to Pandemrix as it was not used after 2010. Our results suggest that the increased NT1 incidence after 2009-2010 pH1N1 is not unique and our study provides an opportunity to develop new hypotheses, e.g., considering other (influenza) viruses or epidemiological events to further investigate the pathophysiology of immune-mediated narcolepsy.

U2 - 10.1093/sleep/zsaa172

DO - 10.1093/sleep/zsaa172

M3 - Article

C2 - 32909046

JO - Sleep

JF - Sleep

SN - 0161-8105

ER -

New 2013 incidence peak in childhood narcolepsy: more than vaccination?

Abstract

Access to Document

Fingerprint

Cite this