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Study Objectives: <p>Sleep disorders and/or disordered sleep represent common clinical presentations of pediatric acute-onset neuropsychiatric syndrome, occurring in up to 80% of affected children, with rapid eye movement (REM) sleep motor disinhibition being a prevalent feature. To date, limited polysomnographic studies have been conducted. Therefore, the objective of this study was to evaluate the polysomnographic characteristics of a cohort of children with pediatric acute-onset neuropsychiatric syndrome, focusing particularly on REM sleep without atonia as assessed by the REM atonia index (RAI), and to compare these characteristics with those of a control group.</p> Methods: <p>Sixty-nine patients diagnosed with pediatric acute-onset neuropsychiatric syndrome were consecutively recruited who underwent comprehensive polysomnographic examinations following standard criteria. Chin muscle tone during REM sleep was evaluated using RAI. Forty-four healthy individuals, matched for age and sex, were used as controls.</p> Results: <p>RAI was significantly lower in patients compared to controls. In controls, RAI showed a significant progressive increase with age from preschool to adolescence. Conversely, this age-related trend was absent in patients, with no significant correlation between RAI and age, with a random distribution of values. Only 1 control individual (1/43) but 25 patients (25/57) exhibited a RAI below the lower limit of the predicted range obtained from controls.</p> Conclusions: <p>Our findings underscore the significance of REM sleep abnormalities in children with pediatric acute-onset neuropsychiatric syndrome. The substantial reduction in RAI and the lack of correlation between RAI and age observed in patients suggest a potential pathological mechanism leading to dysfunction in brainstem structures regulating REM sleep atonia in this population.</p> Citation: <p>Congiu P, Gagliano A, Carucci S, Lanza G, Ferri R, Puligheddu M. REM sleep atonia in patients with pediatric acute-onset neuropsychiatric syndrome: implications for pathophysiology. <i>J Clin Sleep Med</i>. 2025;21(5):757–764.</p>

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REM sleep atonia in patients with pediatric acute-onset neuropsychiatric syndrome: implications for pathophysiology

  • 期刊论文

  • 发表日期：

    2025年5月1日

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  • Patrizia Congiu,
  • Antonella Gagliano,
  • Sara Carucci,
  • Giuseppe Lanza,
  • Raffaele Ferri,
  • Monica Puligheddu

摘要

Study Objectives:

Sleep disorders and/or disordered sleep represent common clinical presentations of pediatric acute-onset neuropsychiatric syndrome, occurring in up to 80% of affected children, with rapid eye movement (REM) sleep motor disinhibition being a prevalent feature. To date, limited polysomnographic studies have been conducted. Therefore, the objective of this study was to evaluate the polysomnographic characteristics of a cohort of children with pediatric acute-onset neuropsychiatric syndrome, focusing particularly on REM sleep without atonia as assessed by the REM atonia index (RAI), and to compare these characteristics with those of a control group.

Methods:

Sixty-nine patients diagnosed with pediatric acute-onset neuropsychiatric syndrome were consecutively recruited who underwent comprehensive polysomnographic examinations following standard criteria. Chin muscle tone during REM sleep was evaluated using RAI. Forty-four healthy individuals, matched for age and sex, were used as controls.

Results:

RAI was significantly lower in patients compared to controls. In controls, RAI showed a significant progressive increase with age from preschool to adolescence. Conversely, this age-related trend was absent in patients, with no significant correlation between RAI and age, with a random distribution of values. Only 1 control individual (1/43) but 25 patients (25/57) exhibited a RAI below the lower limit of the predicted range obtained from controls.

Conclusions:

Our findings underscore the significance of REM sleep abnormalities in children with pediatric acute-onset neuropsychiatric syndrome. The substantial reduction in RAI and the lack of correlation between RAI and age observed in patients suggest a potential pathological mechanism leading to dysfunction in brainstem structures regulating REM sleep atonia in this population.

Citation:

Congiu P, Gagliano A, Carucci S, Lanza G, Ferri R, Puligheddu M. REM sleep atonia in patients with pediatric acute-onset neuropsychiatric syndrome: implications for pathophysiology. J Clin Sleep Med. 2025;21(5):757–764.