Continued Presence of Period Limb Movements During REM Sleep in Patients With Chronic Static Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS)

J Clin Sleep Med. 2018 Jul 15;14(7):1187-1192. doi: 10.5664/jcsm.7222.

Abstract

Study objectives: A major component of pediatric acute-onset neuropsychiatric syndrome (PANS) is disruption of sleep. These disturbances have been reported in the acute phase of diagnosis but it is unknown if these sleep disruptions persist, especially in patients with chronic static symptoms. This retrospective chart review sought to review polysomnography (PSG) tests of patients in whom PANS has been clinically diagnosed in order to assess sleep architecture, periodic limb movements, and presence of rapid eye movement (REM) sleep without atonia (RSWA) after a chronic static course of symptoms, which were refractory to immunomodulatory interventions.

Methods: Patients were retrospectively identified through the PANS clinic at our institution and had to have fully completed a PSG study and be younger than 18 years. PSG with video were reviewed and scored based on established criteria.

Results: We identified 9 patients who met inclusion criteria. The median time from presentation to PSG was 4 years. This study identified PSG-measured periodic limb movement index (PLMI) > 5 events/h in REM sleep in 7 of 9 patients. Two patients with elevated PLMI also demonstrated RSWA, although neither fit a clinical diagnosis of REM sleep behavior disorder. This cohort also demonstrated increased onset of REM sleep (median 134 minutes), insomnia (median total sleep time of 389 minutes), and decreased sleep efficiency (77%).

Conclusions: This study identifies continued sleep disturbances in patients with refractory PANS symptoms several years after diagnosis and treatment. Continued sleep disturbances after presentation and treatment in patients with chronic static PANS may be a contributing factor in prolonged symptomatology of this disease process.

Keywords: REM; pediatric acute-onset neuropsychiatric syndrome; periodic limb movement disorder; periodic limb movements; sleep.

MeSH terms

  • Adolescent
  • Autoimmune Diseases / complications*
  • Child
  • Child, Preschool
  • Chronic Disease
  • Cohort Studies
  • Female
  • Humans
  • Male
  • Obsessive-Compulsive Disorder / complications*
  • Polysomnography / methods
  • REM Sleep Behavior Disorder / complications*
  • REM Sleep Behavior Disorder / diagnosis
  • REM Sleep Behavior Disorder / physiopathology*
  • Retrospective Studies

Supplementary concepts

  • Pediatric acute-onset neuropsychiatric syndrome