Cyclic alternating pattern as a provocative factor in nocturnal paroxysmal dystonia

Abstract

Purpose: We made a polygraphic study of 6 patients with nocturnal paroxysmal dystonia (NPD) in which the cyclic alternating pattern (CAP) parameters were compared with those of a group of age- and sex-matched controls. Methods: All patients met the requirements for NPD diagnosis, characterized by generalized stereotyped movements (dystonic-dyskinetic), with a 1-min centered duration but with no clear evidence of epileptic abnormalities in the waking BEG and during nocturnal recordings. Results: Besides the major events, the NPD polysomnograms also showed shorter, repeated episodes of shorter duration (generally <20 s) consisting of abrupt movements involving one or more body segments. Overall, the motor events in patients with NPD were closely related to periods of unstable non-REM (NREM) sleep, as evidenced by the sequences of CAP, and began during an A phase. According to the conventional scoring parameters, NPD and controls differed only in sleep latency (+14 min in the NPD patients: p < 0.04). However, the architecture of sleep in the group with NPD was characterized by prolonged and irregular NREM/REM cycles. In addition, the NPD recordings showed significantly higher values of CAP rate (p < 0.0001). When major motor attacks were suppressed by medication, sleep was characterized by a decrease in the excessive amounts of CAP rate and by a more regular architecture. Conclusions: The modulatory role of CAP on nocturnal motor events is reported.