ArticleBrain : a journal of neurology2011
Complex movement disorders at disease onset in childhood narcolepsy with cataplexy.
Article in Brain : a journal of neurology, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 60 papers, 1 of them a synthesis that pooled it.
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Who cites it
60 citing papers in PubMed, 1 synthesis or guideline pooled it, 198 citations in OpenAlex.
- Narcolepsy and psychosis: A systematic review.Acta psychiatrica Scandinavica · 2021Pooled it
- Missense Variants in the A Isoform of FGF13 as a Novel Cause of Paroxysmal Dyskinesia.Movement disorders : official journal of the Movement Disorder Society · 2026Article
- Age at Onset and Delays in Diagnosis of Central Disorders of Hypersomnolence Over the Past 30 Years.Journal of sleep research · 2026Article
- Narcolepsy with co-occurring epilepsy: diagnostic pitfalls and management strategy.Therapeutic advances in neurological disorders · 2026Article
- The role of histamine in narcolepsy type 1: a narrative review.Frontiers in neurology · 2026Review
- Present and Future of Central Disorders of Hypersomnolence.Journal of sleep research · 2025Review
- Narcolepsy and rapid eye movement sleep.Journal of sleep research · 2025Review
- Narcolepsy: Beyond the Classic Pentad.CNS drugs · 2025Review
- Understanding the Patient Experience with Twice-Nightly Sodium Oxybate Therapy for Narcolepsy: A Social Listening Experiment.Brain sciences · 2024Article
- Role of Daytime Continuous Polysomnography in the Diagnosis of Pediatric Narcolepsy Type 1.Neurology · 2024Article
- Idling for Decades: A European Study on Risk Factors Associated with the Delay Before a Narcolepsy Diagnosis.Nature and science of sleep · 2022Article
- Orexin/Hypocretin System Dysfunction in ESSENCE (Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations).Neuropsychiatric disease and treatment · 2022Article
- The Genetics of Sleep Disorders in Children: A Narrative Review.Brain sciences · 2021Review
- Narcolepsy type 1 features across the life span: age impact on clinical and polysomnographic phenotype.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2021Article
- Hypothalamus and amygdala functional connectivity at rest in narcolepsy type 1.NeuroImage. Clinical · 2021Article
- Commentary on the Paper by Gagliano et al: Artificial Neural Networks Analysis of Polysomnographic and Clinical Features in Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS): From Sleep Alteration to "Brain Fog" [Letter].Nature and science of sleep · 2021Article
- Recognizing the Symptom Spectrum of Narcolepsy to Improve Timely Diagnosis: A Narrative Review.Nature and science of sleep · 2021Review
- The Impacts of Age and Sex in a Mouse Model of Childhood Narcolepsy.Frontiers in neuroscience · 2021Article
- Early detection of Niemann-pick disease type C with cataplexy and orexin levels: continuous observation with and without Miglustat.Orphanet journal of rare diseases · 2020Article
- Clinical Characteristics of Cataplectic Attacks in Type 1 Narcolepsy.Current neurology and neuroscience reports · 2020Review
Corrections and comments
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Authors and funding
14 authors at 8 institutions in 4 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Narcolepsy with cataplexy is characterized by daytime sleepiness, cataplexy (sudden loss of bilateral muscle tone triggered by emotions), sleep paralysis, hypnagogic hallucinations and disturbed nocturnal sleep. Narcolepsy with cataplexy is most often associated with human leucocyte antigen-DQB1*0602 and is caused by the loss of hypocretin-producing neurons in the hypothalamus of likely autoimmune aetiology. Noting that children with narcolepsy often display complex abnormal motor behaviours close to disease onset that do not meet the classical definition of cataplexy, we systematically analysed motor features in 39 children with narcolepsy with cataplexy in comparison with 25 age- and sex-matched healthy controls. We found that patients with narcolepsy with cataplexy displayed a complex array of 'negative' (hypotonia) and 'active' (ranging from perioral movements to dyskinetic-dystonic movements or stereotypies) motor disturbances. 'Active' and 'negative' motor scores correlated positively with the presence of hypotonic features at neurological examination and negatively with disease duration, whereas 'negative' motor scores also correlated negatively with age at disease onset. These observations suggest that paediatric narcolepsy with cataplexy often co-occurs with a complex movement disorder at disease onset, a phenomenon that may vanish later in the course of the disease. Further studies are warranted to assess clinical course and whether the associated movement disorder is also caused by hypocretin deficiency or by additional neurochemical abnormalities.
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