Clinical, behavioural and polysomnographic correlates of cataplexy in patients with narcolepsy/cataplexy

Mattarozzi, Katia; Bellucci, Claudia Crilly; Campi, Claudio; Cipolli, Carlo; Ferri, Raffaele; Franceschini, Christian; Mazzetti, Michela; Russo, Paolo Maria; Vandi, Stefano; Vignatelli, Luca; Plazzi, Giuseppe

doi:10.1016/j.sleep.2007.05.006

Cited by 78 publications

(37 citation statements)

References 24 publications

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“…Cataplexy, a symptom characterized by sudden and brief (5–30 s) losses of muscle tone (partial or total) triggered by emotions such as laughter and surprise, is considered pathognomonic of narcolepsy with cataplexy (American Academy of Sleep Medicine, 2005; Vetrugno et al ., 2010). Additionally, nocturnal sleep in narcolepsy with cataplexy is not only fragmented by frequent awakenings, but also disturbed by symptoms reflecting abnormal motor control during sleep, such as periodic and non-periodic limb movements, restless legs syndrome and rapid eye movement (REM) sleep behaviour disorder (Ferri et al ., 2006, 2008; Dauvilliers et al ., 2007 b ; Mattarozzi et al ., 2008; Knudsen et al ., 2010; Plazzi et al ., 2008 a , 2010). …”

Section: Introductionmentioning

confidence: 99%

Complex movement disorders at disease onset in childhood narcolepsy with cataplexy

Plazzi

Pizza

Palaia

et al. 2011

Brain

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166

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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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Section: Introductionmentioning

confidence: 99%

Complex movement disorders at disease onset in childhood narcolepsy with cataplexy

Plazzi

Pizza

Palaia

et al. 2011

Brain

Self Cite

166

137

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“…There is no evidence that RBD associated with narcolepsy represents a risk factor for the development of neurodegenerative conditions. [33][34][35][36][37] 4) Eating disorders The prevalence of compulsive eating and restrictive eating behaviors is greater in the population with narcolepsy than in the general population. 38 The symptoms of eating disorders do not correlate with medication use or with affective symptoms.…”

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confidence: 99%

Diretrizes brasileiras para o diagnóstico de narcolepsia

Alóe

Alves

Araújo

et al. 2010

Rev. Bras. Psiquiatr.

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Este artigo relata as conclusões da reunião de consenso com médicos especialistas sobre diagnóstico de narcolepsia baseada na revisão dos artigos sobre narcolepsia listados no Medline entre 1980 e 2010. A narcolepsia é uma doença crônica de início entre a primeira e segunda décadas de vida do indivíduo. Os sintomas essenciais são cataplexia e sonolência excessiva. A cataplexia é definida como episódios súbitos, recorrentes e reversíveis de fraqueza da musculatura esquelética desencadeados por situações de conteúdo emocional. Os sintomas acessórios são alucinações hipnagógicas, paralisia do sono e sono fragmentado. Critérios de diagnóstico clínico de acordo com a Classificação Internacional dos Transtornos do Sono são de sonolência excessiva e cataplexia. Recomenda-se a realização de polissonografia seguida do teste de latência múltipla do sono em um laboratório de sono para confirmação e diagnóstico de comorbidades. Quando não houver cataplexia, deve haver duas ou mais sonecas com sono REM no teste de latência múltipla do sono. Tipagem HLA-DQB1*0602 positiva com níveis de hipocretina-1 abaixo de 110pg/mL devem estar presentes para o diagnóstico de narcolepsia sem cataplexia e sem sonecas com sono REM.

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“…12 However, it is not clear if this was performed in other studies using visual methods of scoring as well, as this was not reported…”

Section: 2829mentioning

confidence: 98%

“…10 RBD and/or RSWA are thought to be common in narcolepsy with cataplexy and rare in narcolepsy without cataplexy. 11,12 It has been proposed that hypocretin defi ciency plays an essential role for both RBD and cataplexy in narcolepsy.…”

Section: S C I E N T I F I C I N V E S T I G a T I O N Smentioning

confidence: 99%

Loss of Rapid Eye Movement Sleep Atonia in Patients with REM Sleep Behavioral Disorder, Narcolepsy, and Isolated Loss of REM Atonia

Khalil

Wright

Walker

et al. 2013

Journal of Clinical Sleep Medicine

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Study Objectives: To compare the amounts of REM sleep without atonia (RSWA) between patients with REM sleep behavior disorder (RBD), "isolated loss of REM atonia," narcolepsy, and control subjects and determine if there were threshold values for the amount of RSWA that differentiate each group from controls. Methods: Retrospective analyses of polysomnography (PSG) records were used employing strict quantitative criteria for the measurement of phasic and tonic EMG activity during REM sleep. The PSG recordings of 47 individuals were analyzed (RBD 16, isolated loss of REM atonia 11, narcolepsy 10, control 10). Results: Patients with the diagnosis of isolated loss of REM atonia had signifi cantly lower levels of EMG activity during REM sleep than those with RBD but higher than control subjects. RSWA was higher in narcolepsy than in loss of REM atonia but lower than for RBD patients. Receiver operating characteristic (ROC) curves provided threshold values with high specifi city and sensitivity in all patient groups with a cutoff value ≥ 1.22% (100% correctly classifi ed) for phasic and ≥ 3.17% for tonic (92% correctly classifi ed) EMG activity in RBD. Conclusion S C I E N T I F I C I N V E S T I G A T I O N SR EM sleep behavior disorder (RBD) is a parasomnia characterized by loss of normal muscle atonia during REM sleep with abnormal motor activity that is proposed to be associated with dream content.1 RBD is associated with neurodegenerative disorders and in particular with synucleinopathies such as Parkinson disease (PD), dementia with Lewy bodies (DLB), and multiple system atrophy (MSA) (secondary RBD). Long-term follow-up studies have shown that a large proportion (38% to 65%) of patients with presumed idiopathic RBD develop Parkinsonism and/or dementia.2,3 EEG and neuropsychological tests have also revealed early evidence of CNS dysfunction in idiopathic RBD patients.4-7 Furthermore, diffusion tensor imaging has uncovered microstructural abnormalities in the brain areas responsible for REM sleep regulation in idiopathic RBD patients. In addition to neurodegenerative disease, RBD is also associated with other sleep disorders. Periodic limb movements of sleep (PLMS) are highly prevalent (70%) in patients with RBD.5 In addition, RBD is frequently seen in patients with narcolepsy.9,10 REM sleep without atonia (RSWA) in narcolepsy was fi rst described in 1992.10 RBD and/or RSWA are thought to be common in narcolepsy with cataplexy and rare in narcolepsy without cataplexy. 11,12It has been proposed that hypocretin defi ciency plays an essential role for both RBD and cataplexy in narcolepsy. Department of Clinical and Experimental Epilepsy, Institute of Neurology, UCL, London UKIsolated loss of REM atonia or RSWA is a condition usually identifi ed incidentally in patients during polysomnography. The loss of REM atonia is an electrophysiological fi nding without accompanying abnormal motor behavior during REM sleep. Although the condition may represent "subclinical" or "preclinical" RBD in some cases, 14 there are no ...

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Clinical, behavioural and polysomnographic correlates of cataplexy in patients with narcolepsy/cataplexy

Cited by 78 publications

References 24 publications

Complex movement disorders at disease onset in childhood narcolepsy with cataplexy

Complex movement disorders at disease onset in childhood narcolepsy with cataplexy

Diretrizes brasileiras para o diagnóstico de narcolepsia

Loss of Rapid Eye Movement Sleep Atonia in Patients with REM Sleep Behavioral Disorder, Narcolepsy, and Isolated Loss of REM Atonia

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