Paroxysmal Nocturnal Dystonia Presenting as Excessive Daytime Somnolence

Maccario, Micheline; Lustman, Lilian I.

doi:10.1001/archneur.1990.00530030061017

Cited by 19 publications

(4 citation statements)

References 12 publications

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“…Two case reports (Oldani et al, 1998b; Nobili et al, 2003) described three patients with several seizures per night with EDS diagnosed according to the ESS scale (scores ranging from 12 to 18), with remission of EDS after successful drug (Oldani et al, 1998b) or surgical (Nobili et al, 2003) treatment of epilepsy. Moreover, Maccario and Lustman (1990) found a pathological level of EDS diagnosed by MSLT (mean sleep latency < 5 min) in two out of four patients. One of these reverted to normal MSLT values after treatment with valproate.…”

Section: Discussionmentioning

confidence: 87%

“…Nocturnal frontal lobe epilepsy (NFLE) (Oldani et al, 1998a; Provini et al, 1999), characterized by complex motor behavior during sleep, could be a model to investigate the influence of sleep fragmentation induced by nocturnal seizures on daytime vigilance. Daytime sleepiness‐related symptoms and disturbed sleep quality have been reported in these patients (Peled and Lavie, 1986; Maccario and Lustman, 1990; Oldani et al, 1996, 1998a, 1998b; Schwalen and Jorg, 1998; Zucconi et al, 2000; Nobili et al, 2003), but without application of validated tools for measurement of sleepiness and comparison with healthy subjects.…”

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confidence: 99%

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Excessive Daytime Sleepiness and Subjective Sleep Quality in Patients with Nocturnal Frontal Lobe Epilepsy: A Case‐Control Study

et al. 2006

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Summary:Purpose: Nocturnal frontal lobe epilepsy (NFLE) may be associated with sleep fragmentation and reduced sleep efficiency. Daytime sleepiness and disturbed sleep quality have been reported in some patients. We investigated the occurrence of daytime sleepiness-related symptoms and subjective sleep quality in patients with NFLE in comparison with matched controls.Methods: Patients with NFLE at a single epilepsy center and matched controls randomly selected from the general population self-administered questionnaires on daytime sleepiness-related symptoms and subjective sleep quality [Epworth sleepiness scale (ESS), Bologna questionnaire on sleepiness-related symptoms (BQS), Berlin questionnaire].Results: Thirty-three patients with NFLE and 27 controls were enrolled. "Tiredness after awakening" and "spontaneous mid-sleep awakenings" were more frequent in NFLE patients than in controls (36.4% versus 11.1%, p = 0.04, and 50.0% versus 22.2%, p = 0.03). The frequency of excessive daytime sleepiness (EDS) did not differ between groups. Posthoc analysis showed that patients with a complaint of "midsleep awakenings" had higher ESS and BQS scores than those without (7.3 versus 4.3, p = 0.004, and 5.0 versus 2.2, p = 0.001, respectively) and more frequent "tiredness after awakening" (56.3% versus 18.8%, p = 0.03).Conclusions: Patients with NFLE have no pathological level of EDS with respect to controls. However, daytime sleepinessrelated symptoms could be more frequent in a subgroup of patients with subjective disturbed sleep quality, irrespective of the perceived frequency of seizures.

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Section: Discussionmentioning

confidence: 87%

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confidence: 99%

Excessive Daytime Sleepiness and Subjective Sleep Quality in Patients with Nocturnal Frontal Lobe Epilepsy: A Case‐Control Study

et al. 2006

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“…Likewise, the 1990 classification of sleep disorders included NPD within the parasomnias [1]. Other authors also concluded that this condition could not be an epileptic disorder [13‐15], some stressing the evidence militating in favor of a parasomnia, similar to night terrors [5, 16]. A psychogenic explanation for this condition was also advanced [17].…”

Section: Discussionmentioning

confidence: 99%

Nocturnal paroxysmal dystonia due to a subfrontal cortical dysplasia

Lombroso

2000

Epileptic Disorders

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The nature and nosology of nocturnal paroxysmal dystonia (NPD) have been controversial. Some authors consider it as a type of parasomnia, akin to night terrors and the official Classification of Sleep Disorder includes NPD within the parasomnias [1]. Others have opined for its epileptic nature, although mainly on circumstantial evidence. The location of the epileptogenic area has been so far unknown. A child with NPD was studied extensively, and in spite of normal scalp EEGs, all of his attacks were shown to originate from his right orbitofrontal cortex. Surgical ablation of an unsuspected cortical dysplastic lesion led to full control.

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“…Seizure frequency in SHE patients can be very high, ranging from one to more than 50 attacks per night (Scheffer et al 1994;Provini et al 1999) and paroxysmal arousals or minor motor events may be even more frequent. SHE patients can complain of nocturnal sleep disruption with spontaneous midsleep awakening, sleep inertia in the morning, tiredness at awakening and excessive sleepiness (Peled and Lavie 1986;Maccario and Lustman 1990;Schwalen and Jorg 1998;Zucconi et al 2000;Alanis-Guevara et al 2005) impacting negatively on their quality of life. Although daytime sleepinessrelated symptoms could be common in SHE patients, the frequency of excessive daytime sleepiness did not statistically differ compared to controls (Vignatelli et al 2006).…”

Section: Clinical Featuresmentioning

confidence: 99%

Sleep related hyper motor epilepsy (SHE): a unique syndrome with heterogeneous genetic etiologies

Bisulli

Licchetta

Tinuper

2019

Sleep Science Practice

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Sleep-related hypermotor epilepsy (SHE), formerly known as Nocturnal Frontal Lobe Epilepsy is a focal epilepsy characterized by seizures with complex hyperkinetic automatisms and/or asymmetric tonic/dystonic posturing occurring mostly during sleep. SHE is a rare disease with an estimated minimum prevalence of 1.8/100,000 individuals and represent about 10% of drug-resistant surgical cases. This disorder, though uncommon, is of considerable interest to a broad spectrum of specialists, from child neurologists to neurosurgeons. Distinguishing this condition from nonepileptic paroxysmal behaviour occurring physiologically or pathologically during sleep is often difficult and sometimes impossible on clinical grounds alone, even for experienced epileptologists and sleep physicians. Recognized aetiologies of SHE are heterogeneous and include acquired injuries, genetic causes and structural anomalies such as focal cortical dysplasia. Multiple aetiologies (structural-genetic) are also possible. Non-specific clinical features distinguished different aetiologies even if SHE due to structural lesions usually manifests with early-onset drug-resistant seizures and showed a worse long-term prognosis. The causative genes for SHE are multiple and encode for proteins involved in different molecular pathways. The cholinergic system and the mTOR pathway are the most relevant. This review will provide an exhaustive overview of the genetic background of SHE.

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Paroxysmal Nocturnal Dystonia Presenting as Excessive Daytime Somnolence

Cited by 19 publications

References 12 publications

Excessive Daytime Sleepiness and Subjective Sleep Quality in Patients with Nocturnal Frontal Lobe Epilepsy: A Case‐Control Study

Excessive Daytime Sleepiness and Subjective Sleep Quality in Patients with Nocturnal Frontal Lobe Epilepsy: A Case‐Control Study

Nocturnal paroxysmal dystonia due to a subfrontal cortical dysplasia

Sleep related hyper motor epilepsy (SHE): a unique syndrome with heterogeneous genetic etiologies

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