Decreased sleep quality and increased sleep related movements in patients with Tourette's syndrome

Cohrs, Stefan; Rasch, T; Altmeyer, S; Kinkelbur, Jörg; Kostanecka, Tatiana; Rothenberger, Aribert; Rüther, Eckart; Hajak, Goeran

doi:10.1136/jnnp.70.2.192

Cited by 103 publications

(79 citation statements)

References 27 publications

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“…The remaining 13 patients had chronic motor or vocal tic disorders Vandenbergh et al, 2000;Comings, 2001]. Many TS patients also have a chronic history of sleep disorders and ADHD comorbidity [Allen et al, 1992;Freeman et al, 2000;Cohrs et al, 2001].…”

Section: Clinical Assessmentmentioning

confidence: 99%

“…In particular, a rare case of autosomal recessive canine narcolepsy arising from a Glu54Lys mutation of the OX2R gene suggested the value of studying orexin/hypocretin system genes in sleep disorders: including those comorbid with ADHD [Allen et al, 1992;Freeman et al, 2000;Cohrs et al, 2001].…”

Section: Introductionmentioning

confidence: 99%

“…In particular, there is evidence that sleep disturbances diagnosed as excessive daytime sleepyness (EDS) [Carmelli et al, 2001] or narcolepsy [Taheri and Mignot, 2002] has a genetic component. Poor quality of sleep may also be involved in disorders such as attention deficit-hyperactivity disorder (ADHD) comorbid with Tourette syndrome (TS) [Allen et al, 1992;Freeman et al, 2000;Cohrs et al, 2001;Kostanecka-Endress et al, 2003] that may have complex genetic origins [Barr and Sandor, 1998;Comings, 2001].…”

Section: Introductionmentioning

confidence: 99%

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Variants of the orexin2/hcrt2 receptor gene identified in patients with excessive daytime sleepiness and patients with Tourette's syndrome comorbidity

Thompson

Comings²,

Abu-Ghazalah

et al. 2004

American J of Med Genetics Pt B

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The orexin-2/hypocretin-2 (OX2R) receptor gene is mutated in canine narcolepsy and disruption of the prepro-orexin/hypocretin ligand gene results in both an animal model of narcolepsy and sporadic cases of the human disease. This evidence suggests that the structure of the OX2R gene, and its homologue, the OX1R gene, both members of the G protein-coupled receptor (GPCR) family, and the gene encoding the peptide ligands, the prepro-orexin/hypocretin gene, may be variables in the etiology of sleep disorders. We report a single stranded conformational polymorphism (SSCP) analysis of the coding regions of these genes in idiopathic sleep disorder patients diagnosed with excessive daytime sleepiness (EDS) (n = 28), narcolepsy (n = 28), Tourette's syndrome/chronic vocal or motor tic disorder (n = 70), and control subjects (n = 110). Two EDS patients showed a Pro11Thr change. One Tourette's syndrome patient was found to have a Pro10Ser alteration. The Pro10Ser and Pro11Thr variants were not found in non-disease populations. Analysis of the ability of the mutant receptors to mobilize calcium compared to the wild-type receptor in response to orexin agonists indicated that they resulted in decreased potency at high (etaM) concentrations of orexin ligands. Further work is warranted to study the variability of the orexin/hypocretin system in a variety of disorders characterized by EDS.

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Section: Clinical Assessmentmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Variants of the orexin2/hcrt2 receptor gene identified in patients with excessive daytime sleepiness and patients with Tourette's syndrome comorbidity

Thompson

Comings²,

Abu-Ghazalah

et al. 2004

American J of Med Genetics Pt B

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“…[12][13][14] There is a similar dearth of pediatric PSG studies in neuropsychiatric disorders, particularly with regard to documenting motor-related NREM/REM abnormalities and RBD symptoms. However, the literature suggests various REM abnormalities are present across many disorders.…”

Section: Brief Summarymentioning

confidence: 99%

Rapid Eye Movement Sleep Abnormalities in Children with Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS)

Gaughan

Buckley

Hommer

et al. 2016

Journal of Clinical Sleep Medicine

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Study Objectives: Polysomnographic investigation of sleep architecture in children presenting with pediatric acute-onset neuropsychiatric syndrome (PANS). Methods: Fifteen consecutive subjects meeting criteria for PANS (mean age = 7.2 y; range 3-10 y) underwent single-night full polysomnography (PSG) read by a pediatric neurologist. Results: Thirteen of 15 subjects (87%) had abnormalities detected with PSG. Twelve of 15 had evidence of rapid eye movement (REM) sleep motor disinhibition, as characterized by excessive movement, laughing, hand stereotypies, moaning, or the continuation of periodic limb movements during sleep (PLMS) into REM sleep. Conclusions: This study shows various forms of REM sleep motor disinhibition present in a population of children with PANS. Keywords: obsessive compulsive disorder, PANS, polysomnography, REM sleep behavior disorder Citation: Gaughan T, Buckley A, Hommer R, Grant P; Williams K, Leckman JF, Swedo SE. Rapid eye movement sleep abnormalities in children with pediatric acute-onset neuropsychiatric syndrome (PANS). J Clin Sleep Med 2016;12(7):1027-1032. I NTRO DUCTI O N Schenck et al.1 were the first to suggest that rapid eye movement (REM) sleep neurobehavioral disorders were a separate category of parasomnia in a 1986 case series of four males aged 67-72 y. Polysomnography (PSG) of these four subjects showed a loss of chin atonia and high limb-twitch activity among other REM sleep pathology, whereas videography showed various REM sleep behaviors including punching, kicking, and dream enactment.1 Since then, REM sleep behavior disorder (RBD) has been recognized across a wide range of ages, sometimes decades prior to the onset of other neurobehavioral changes and often heralding serious neurodegenerative conditions affecting synuclein.2 A recent retrospective study of patients showing REM sleep without atonia on PSG found a majority (73%) of patients had idiopathic RBD, defined as RBD in the absence of known neurological or sleep disorders.3 Recent reports suggest that clinical and subclinical forms of RBD occur in children and adolescents.4 Although pediatric RBD is considered to be a rare occurrence, its prevalence may be underestimated due to limited awareness, overlap with other parasomnias, and the requirement of nocturnal PSG for diagnosis.4,5 Additionally, it is unclear whether or not the adult clinical definitions should pertain to pediatric presentations due to a current lack of neuropathologic studies of childhood RBD.4 Differentiating RBD from other parasomnias in the younger population is significant clinically because of the potential to inform on neuropsychiatric state and therefore guide treatment choices, as evidenced in conditions such as narcolepsy where 60% of patients will have RBD. In adult psychiatric populations, RBD has been associated with the use of psychotropic medications and particularly with serotonin reuptake-blocking antidepressants.3,7 However, a recent study suggests that antidepressants cannot fully explain RBD symptomatology in their psychiat...

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“…[8][9][10][11] Some researchers also reported increased general movements in REM sleep. 12 Previously, there was no report of REM sleep behaviour disorder (RBD) in this population, and only two single case history reports existed 13,14 describing RBD in children, until our initial description of a series of pre-pubescent patients with Tourette's syndrome 15 and a report of RBD in otherwise asymptomatic children.…”

mentioning

confidence: 99%

REM Sleep Behaviour Disorder in a Child with Tourette's Syndrome

Trajanović

Voloh

Shapiro

et al. 2004

Can. j. neurol. sci.

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572A number of studies have documented a variety of sleep disorders associated with To u r e t t e 's syndrome, 1 -6 i n c l u d i n g higher incidence of parasomnias, and periodic limb movement disorder 7,8 persisting both in rapid eye movement (REM) and non-REM sleep. [8][9][10][11] Some researchers also reported increased general movements in REM sleep. 12 Previously, there was no report of REM sleep behaviour disorder (RBD) in this population, and only two single case history reports existed 13,14 describing RBD in children, until our initial description of a series of pre-pubescent patients with Tourette's syndrome 15 and a report of RBD in otherwise asymptomatic children. 16 In our previous work 15 we presented a series of 16 patients with To u r e t t e 's syndrome, age 6-65 (14 males) who were referred to our clinic for a variety of sleep-related complaints, but predominantly for suspected nocturnal movement disorder (periodic leg movements in sleep (PLMS), restless legs syndrome (RLS), nocturnal tics) and parasomnic behaviour. In 12 patients we observed raised muscle activity in REM sleep, either in the form of (periodic) leg movements or attenuated tics, or (majority) as augmented phasic electromyograph (EMG) without associated movements. Three of these patients had confirmed, and two strongly possible RBD. Following the initial series of patients, further patients with the various sleep pathology were seen, including the case presented in this article. CASE HISTORYWe present a case of a seven-year-old boy with Tourette's syndrome and RBD. The patient was seen over the four-year period starting in 1998 when he was three years old. Polysomnographic (PSG) sleep assessments were done in late 1999 and mid-2002. ABSTRACT: Purpose:To describe an association of Tourette's syndrome with rapid eye movement sleep behaviour disorder (RBD) in a prepubescent boy. Methods: A four year longitudinal single-case s t u d y. R e s u l t s : The co-existence of To u r e t t e 's syndrome and RBD was confirmed after polysomnographic studies using the standard criteria. The authors propose possible overlap in the pathophysiological mechanisms underlying the two disorders. Can.

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Decreased sleep quality and increased sleep related movements in patients with Tourette's syndrome

Cited by 103 publications

References 27 publications

Variants of the orexin2/hcrt2 receptor gene identified in patients with excessive daytime sleepiness and patients with Tourette's syndrome comorbidity

Variants of the orexin2/hcrt2 receptor gene identified in patients with excessive daytime sleepiness and patients with Tourette's syndrome comorbidity

Rapid Eye Movement Sleep Abnormalities in Children with Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS)

REM Sleep Behaviour Disorder in a Child with Tourette's Syndrome

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