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.
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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...