Valentina Gnoni, Nazanin Biabani, Istvan Papp, Camilla Speicher, Adam Birdseye, Sakina Dastagir, Martina Mulas, Monica Puligheddu, Clive Ballard, Luigi Ferini-Strambi, David O'Regan, Ivana Rosenzweig
In this case, longitudinal RSWA did not behave as a single monotone scalar. Component-wise reporting alongside explicit REM sleep opportunity and exclusion accounting may be essential for interpreting apparent longitudinal change and relating electrophysiology to behavioral expression.
BACKGROUND: Quantitative REM sleep without atonia (RSWA) metrics are increasingly used to characterize isolated REM sleep behavior disorder (iRBD) and as candidate biomarkers. Cohort follow-up often reports rising RSWA, but within-individual trajectories, particularly when tonic and phasic components diverge, remain sparsely documented.
METHODS: A man in his mid-seventies with iRBD underwent four attended video-polysomnograms over -6.5 years. Submental EMG was quantified using a REM sleep atonia index (RAI) with pre-specified exclusion of REM mini-epochs contaminated by artifact or physiological activation (arousal-linked activation and respiratory-event adjacency). Tonic and phasic submental RSWA components were extracted concurrently. Diary-anchored interviews around each visit captured enactment frequency, ordinal ratings of dream vividness/recall and sleep-related accidental harm.
RESULTS: Corrected RAI varied non-monotonically (0.268, 0.401, 0.313, 0.463). Tonic and phasic RSWA dissociated: tonic activity declined overall, whereas phasic activity rose early and remained elevated. PSG-night enactment frequency tracked REM sleep opportunity (Spearman ρ = 0.89; n = 4) more closely than corrected RAI or tonic/phasic indices. Sleep-related harm was absent at the final visit despite persistent phasic activity.
CONCLUSION: In this case, longitudinal RSWA did not behave as a single monotone scalar. Component-wise reporting alongside explicit REM sleep opportunity and exclusion accounting may be essential for interpreting apparent longitudinal change and relating electrophysiology to behavioral expression.