scholarly journals 528 REM Sleep Without Atonia in Idiopathic REM Sleep Behavior Disorder in the North American Prodromal Synucleinopathy Cohort

SLEEP ◽  
2021 ◽  
Vol 44 (Supplement_2) ◽  
pp. A208-A208
Author(s):  
John Feemster ◽  
Paul Timm ◽  
Luke Teigen ◽  
Sarah Faber ◽  
Tyler Steele ◽  
...  

Abstract Introduction Idiopathic/isolated REM sleep behavior disorder (iRBD) is a prodromal alpha-synucleinopathy characterized by dream enactment behavior and REM sleep without atonia (RSWA). We sought to define quantitative RSWA diagnostic thresholds in the North American Prodromal Synucleinopathy (NAPS) Consortium cohort. We analyzed RSWA between iRBD patients across participating NAPS sleep centers, compared to normative controls, and hypothesized that previous diagnostic RSWA thresholds were overestimates. Methods All digital polysomnography files were converted to European Data Format and scored at a central laboratory (Mayo Clinic) which standardized display scoring montages, channel sensitivities, and filtering, and scripted computational analyses for visual scoring. RSWA was quantitatively analyzed in the submentalis (SM) and anterior tibialis (AT) muscles in iRBD (n=86) patients and controls (n=118) utilizing well validated visual (Mayo) and automated (RAI) methods. Parametric statistics were used to compare RSWA metrics, and RSWA thresholds were developed using receiver operating characteristic curves. Results RSWA was significantly higher for the RAI and all visual individual and combined muscle activity metrics in iRBD compared to controls (all p<0.001). Average SM phasic measures were: 14.2% (Mayo), 17.9% (McGill), 18.5% (UCLA), and 9.4% (Washington University). Average AT phasic measures at each site were: 26.7% (Mayo), 17.1% (McGill), 23.3% (UCLA), and 17.4% (Washington University). Average SM/AT ‘any’ measures at each site were: 45.4% (Mayo), 35.9% (McGill), 53.4% (UCLA), and 23.5% (Washington University). Overall cohort RBD diagnostic thresholds (AUC, specificity/sensitivity) were: SM phasic 4.9% (90.0, 82.2%/83.7%); AT phasic 7.6% (88.7%, 82.2%/81.4%) and combined SM/AT ‘any’ 13% (94.6, 83.9%/96.5%). Conclusion RSWA thresholds in the NAPS cohort were substantially lower than previously reported, suggesting previously overestimated diagnostic RSWA thresholds due to smaller, enriched patient samples and overfit statistical modeling. Confirmation of these findings in the complete NAPS cohort (n=300 iRBD patients across all 10 NAPS centers) is planned. Support (if any):

2021 ◽  
Vol 2 (Supplement_1) ◽  
pp. A46-A46
Author(s):  
D Levendowski ◽  
J Lee-Iannotti ◽  
D Shprecher ◽  
C Guevarra ◽  
P Timm ◽  
...  

Abstract Purpose Compare agreements between polysomnography-based (PSG) diagnosis of isolated REM-sleep-behavior-disorder (iRBD) and Non-REM-Hypertonia (NRH), a novel biomarker independently associated with synucleinopathy-related neurodegenerative diseases. Methods Sixteen patients with histories of dream-enactment-behavior (DEB)(women=38%; age:64.6±13.0) underwent PSG with simultaneously-recorded Sleep Profiler (SP). Two boarded sleep neurologists independently characterized iRBD. Physician1 combined abnormal qualitative REM-sleep-without-atonia (RSWA) by submental electromyography, with video-confirmation of probably DEB. Physician2 relied solely on qualitative RSWA. SP was auto-staged, technically reviewed, and reprocessed for automated abnormal NRH detection. Kappa scores measured physician and NRH agreements. Results In the 14 records with REM sleep, iRBD was characterized in: Physician1=64%, Physician2=79%, NRH=71% of the records. Across the three methods, unanimous iRBD agreement occurred in 57% of the records (positive=7, negative=1). The between-physician agreement in iRBD classifications was fair (kappa=0.32). The agreement between NRH and Physician1 was moderate (kappa=0.52) versus slight with Physician2 (kappa=0.05). NRH comparisons to consensus physician agreement yielded one false-positive and one false-negative iRBD finding. Physician2 classified: a) iRBD in two cases that were negative by Physician1 and NRH, and b) one negative case that Physician1 and NRH characterized as iRBD. Physician1 identified one negative case that was classified iRBD by Physician2 and NRH. Additionally, NRH was abnormal in one of the two records with no REM sleep. Discussion NRH may assist in iRBD risk assessment, given it agreed with at least one physician in 86% of the cases and the between-physician iRBD agreement was only fair. NRH also characterized iRBD-risk in patients with insufficient REM sleep for RSWA assessment.


Neurology ◽  
2020 ◽  
pp. 10.1212/WNL.0000000000011157
Author(s):  
Elena Antelmi ◽  
Marco Filardi ◽  
Fabio Pizza ◽  
Stefano Vandi ◽  
Monica Moresco ◽  
...  

Objective:The aim was to study the effect of stable treatment with Sodium Oxybate (SO) on nocturnal REM sleep behavior disorder (RBD) and REM sleep without atonia (RSWA) that severely affected children with type 1 narcolepsy (NT1.Methods:Nineteen NT1 children and adolescents (nine females; mean age 12.5±2.7, mean disease duration: 3.4±1.6 years) underwent neurological investigations and video-polysomnography (v-PSG) at baseline and after three months of stable treatment with SO.v-PSG was independently analysed by two sleep experts, in order to rate RBD episodes. RSWA was automatically computed by means of the validated REM sleep atonia index (RAI).Results:Compared to baseline, RAI significantly improved (p< 0.05) and complex movements during REM sleep were remarkably reduced after stable treatment with SO. Compared to baseline, children also reported improvement in clinical complaints and showed a different nighttime sleep stage architecture.Conclusions:RBD and RSWA improved after treatment with SO, pointing to a direct role of the drug in modulating motor control during REM sleep.


SLEEP ◽  
2006 ◽  
Vol 29 (10) ◽  
pp. 1321-1325 ◽  
Author(s):  
Jean-François Gagnon ◽  
Dominique Petit ◽  
Maria Livia Fantini ◽  
Sylvie Rompré ◽  
Serge Gauthier ◽  
...  

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