Table 1.
Individual study data for combination multimodal therapy for dystonia.
| Combination of multiple treatments (n = 29) | ||||||||||
|---|---|---|---|---|---|---|---|---|---|---|
| Author | Study design | Participant characteristics | Intervention | Assessment outcomes | Results | AAN class | ||||
| Active group | Control group | Active | Control | |||||||
| Focal Cervical Dystonia | ||||||||||
| Bleton. et al., 202416 France | Cross-over | N | 5 | Treatment | Motor training + tDCS | tDCS | TWSTRS (severity) | Significantly greater & longer improvement in dystonia severity | III | |
| M/F | 1/4 | Details | Cerebellar anodal tDCS + activating impaired muscles | |||||||
| Age (y) | 63 (40–79) | Frequency/Duration | 20 min/d, 3–5 d/w, 1w | |||||||
| Dec-Ćwiek. et al., 202217 Poland | RCT cross-over | N | 19 | Treatment | Kinesiotaping + BoNT | Sham taping + BoNT & BoNT alone | TWSTRS, CDQ24 (severity, subjective) | Significant improvement in QoL No significant difference in dystonia severity | II | |
| M/F | 4/15 | Details | Kinesiotaping applied to shoulder Kinesiotaping initiated 7 d after BoNT | |||||||
| Age (y) | 54 ± 12 | Frequency/Duration | 3 conditions; 12w/condition; 36w | |||||||
| Castagna. et al., 202018 Italy | Cross-over | N | 15 | Treatment | Supervised PT + BoNT | BoNT | TWSTRS (severity) | Significant improvement in dystonia severity with addition of PT | III | |
| M/F | 8/7 | Details | Augmented feedback of movement (visual & acoustic) + BoNT PT initiated just after BoNT | |||||||
| Age (y) | 48 ± 9 | Frequency/Duration | BoNT: 2 sessions + Exercise: 18 sessions, 6w, 18w | |||||||
| de Oliveira. et al., 202028 Brazil | Pre-post | N | 2 | Treatment | Progressive exercises + tDCS | NA | TWSTRS, WCRS (severity, pain) | Improvement in dystonia severity & pain | IV | |
| M/F | 1/1 | Details | Exercises for cervical and trunk muscles + tDCS (2 mA, 20 min, PMC) | |||||||
| Age (y) | 79, 48 | Frequency/Duration | 15 sessions, 3 m | |||||||
| Werner. et al., 201919 Germany | RCT | N | 18 | Treatment | Supervised PT + BoNT | BoNT | ROM, SF-36, TWSTRS (function, subjective, severity) | Significant improvement in ROM, subjective symptoms, & severity with addition of PT | II | |
| M/F | 3/15 | Details | Reduction of pathological movement patterns PT performed between two BoNT injection sessions | |||||||
| Age (y) | 63 ± 13 | Frequency/Duration | BoNT: 2 sessions + PT: 45 min/session, 2/w, 3m | |||||||
| Hu. et al., 201920 USA | RCT | N | 8 | 8 | Treatment | Supervised and home-based PT + BoNT | BoNT | TWSTRS (severity) | Significant improvement in dystonia severity with addition of PT | II |
| M/F | 4/4 | 5/3 | Details | Stretching, range-of-motion, isometric exercises PT initiated after BoNT on the same day | ||||||
| Age (y) | 64 ± 7 | 67 ± 7 | Frequency/Duration | 15 min/d, 5 d/w, 6 w | ||||||
| van den Dool. et al., 201927 Netherlands | RCT | N | 48 | 48 | Treatment | Supervised customized PT + BoNT | Supervised regular PT + BoNT | TWSTRS disability (severity) | Both groups significantly improved in dystonia severity | I |
| M/F | 19/29 | 18/30 | Details | Stretching, range-of-motion, passive mobilization, biofeedback PT initiated 2w after BoNT | ||||||
| Age (y) | 59 ± 9 | 57 ± 9 | Frequency/Duration | 1/w, 1y | ||||||
| Stankovic. et al., 201825 Serbia | RCT | N | 9 | 4 | Treatment | BoNT + PT supervised clinic-based & home-based | Home based PT | Tsui scale, TWSTRS (severity) | Significantly greater & longer improvement in dystonia severity | II |
| M/F | 11/3 | Details | Exercises, stretching, OT, functional therapy PT initiated 5d after BoNT | |||||||
| Age (y) | 42 ± 5 | Frequency/Duration | BoNT: 1 session + Supervised PT: 5 d/w, 2w + Home PT, 6m | |||||||
| Bradnam. et al., 201629 Australia | RCT | N | 16 | Treatment | rTMS + motor training | sham rTMS | TWSTRS, CDQ24 (severity, pain, subjective) | Significant improvement in dystonia severity, pain, subjective symptoms | II | |
| M/F | 6/10 | Details | Intermittent theta-burst stimulation for cerebellum + Motor training for neck | |||||||
| Age (y) | 28–72 | Frequency/Duration | 2s train every 10s, total of 190s or 600 pulses, 10 sessions | |||||||
| Counsell. et al., 201622 UK | RCT | N | 55 | 55 | Treatment | Specialized supervised PT | Relaxation, exercises to increase ROM, core stability (home-based) | TWSTRS (severity) | Both groups significantly improved in severity | I |
| M/F | 17/38 | 13/42 | Details | Strengthening of underactive muscles, advice about posture, awareness of body position, relaxation of overactive muscles | ||||||
| Age (y) | 55 (13) | 57 (12) | Frequency/Duration | 45 min/session, 1 session/w, 24w | ||||||
| Queiroz. et al., 201221 Brazil | Case-control | N | 20 | 20 | Treatment | PT with FES + BoNT | BoNT | TWSTRS, SF-36 (severity, subjective) | Significant improvement in ADL & subjective pain | III |
| M/F | 9/11 | 11/9 | Details | Motor learning exercises, kinesiotherapy, FES on antagonist muscles PT initiated 15 d after BoNT | ||||||
| Age (y) | 52 (14) | 50 (12) | Frequency/Duration | 25 min/session, 5 session/w, 4w | ||||||
| El-Bahrawy. et al., 200926 Egypt | RCT | N | 20 | 20 | Treatment | Exercise therapy & TENS + BoNT | Sham TENS + BoNT | Head posture & ROM, Purdue Peg Board Test (function) | Significant improvement in head posture | II |
| M/F | 13/7 | 12/8 | Details | Stretching, training of voluntary movements, TENS BoNT performed at least 1m before PT | ||||||
| Age (y) | 32 ± 4 | 32 ± 3 | Frequency/Duration | BoNT: 1 session + PT: 3 sessions/w, 18 sessions; 6w | ||||||
| Tassorelli. et al., 200623 Italy | RCT cross-over | N | 20 | 20 | Treatment | Exercise therapy + BoNT | BoNT | Tsui scale, TWSTRS, ADL, total pain scale (severity, subjective, pain) | Both groups significantly improved in dystonia severity Pain & ADL significantly improved in exercise group; BoNT effects prolonged | II |
| M/F | 7/13 | 6/14 | Details | Massage, passive myofascial elongation maneuvers, stretching, biofeedback PT initiated just after BoNT | ||||||
| Age (y) | 50 ± 16 | 52 ± 14 | Frequency/Duration | 60–90 min/d, daily, 2w | ||||||
| Ramdharry. et al., 200624 UK | Case report | N | 1 | Treatment | PT + BoNT | NA | TWSTRS (severity) | Improvement in dystonia severity & increased BoNT effects | IV | |
| M/F | M | Details | Strengthen neck muscles PT initiated just after BoNT | |||||||
| Age (y) | NA | Frequency/Duration | 14 sessions, 6m | |||||||
| Gildenberg. et al., 198130 USA | Pre-post | N | 29 | Treatment | Biofeedback training + TENS | NA | Overall Symptoms (subjective) | 4/29 responded to biofeedback training 3/29 responded to TENS | IV | |
| M/F | NA | Details | EMG biofeedback, relaxation, TENS over sternocleidomastoid | |||||||
| Age (y) | NA | Frequency/Duration | Biofeedback training was tried first TENS performed to ineffective patients | |||||||
| Meige Syndrome and Laryngeal Dystonia | ||||||||||
| Cairns. et al., 199131 Canada | Case report | Type | Meige syndrome | Treatment | Relaxation + Cognitive restructuring + BoNT | NA | Overall Symptoms (subjective) | Improvement in subjective motor symptom | IV | |
| N | 1 | Details | Progressive muscle relaxation + Restructure cognition Timing of BoNT not mentioned | |||||||
| M/F | F | Frequency/Duration | Supervised: 1 session/w, 10w + Home: daily, 1y | |||||||
| Age (y) | Late 40 | |||||||||
| Silverman. et al., 201232 USA | RCT | Type | ADSD | Treatment | Voice training + BoNT | Sham voice training + BoNT & BoNT alone | BoNT effect, voice related QoL (BoNT effect, subjective) | All three groups significantly improved voice-related QoL No significant difference between groups | II | |
| N | 10/10/11 | Details | Voice education, relaxation, laryngeal massage, vocal exercises Voice training initiated 3w after BoNT | |||||||
| M/F | 5/26 | Frequency/Duration | 5 sessions, 12w | |||||||
| Age (y) | 48 (23–78) | |||||||||
| Murry. et al., 199533 USA | Case-control | Type | ADSD | Healthy control | Treatment | Voice therapy + BoNT | BoNT | Air flow rate (function) | Significant improvement in air flow rate Significant prolongation of interval between BoNT injections | III |
| N | 17 | 10 | Details | Voice therapy Voice training initiated within 3w after BoNT | ||||||
| M/F | 3/14 | 1/9 | Frequency/Duration | BoNT: 1 session + Voice therapy: 5 sessions, 9–54 w | ||||||
| Age (y) | 51 (27–74) | 52 (31–71) | ||||||||
| Focal Limb Dystonia | ||||||||||
| de Oliveira. et al., 202028 Brazil | Pre-post | Type | Writer’s cramp | Treatment | Progressive exercises + rTMS | NA | TWSTRS, WCRS (severity, pain) | Improvement in dystonia severity & pain | IV | |
| N | 1 | Details | Exercises for wrists and finger extensor muscles + rTMS (1Hz, 1200 pulses, 80%RMT, premotor cortex) | |||||||
| M/F | F | |||||||||
| Age (y) | 46 | Frequency/Duration | 15 sessions, 3m | |||||||
| Kimberley. et al., 201543 USA | RCT cross-over | Type | Writer’s cramp | Treatment | Sensorimotor rehab + rTMS | Stretching & massage + rTMS | Global rating, arm dystonia disability scale (subjective, severity) | Both groups significantly improved subjective symptoms & dystonia severity. No significant difference between groups | II | |
| N | 9 | Details | Learning based sensorimotor rehab + rTMS (1Hz, 80% RMT, 1200 pulses, premotor cortex) | |||||||
| M/F | 6/3 | Frequency/Duration | 5d | |||||||
| Age (y) | 46 ± 10 | |||||||||
| Rosset-Llobet. et al., 201534 Spain | RCT | Type | Musician’s dystonia | Treatment | Sensorimotor rehab + tDCS | Sensorimotor rehab + sham tDCS | Dystonia severity score (severity) | Both groups significantly improved dystonia severity Adding tDCS showed significantly greater improvement | I | |
| N | 30 | Details | Splints to fingers with compensatory movements + tDCS (2mA to parietal cortex) | |||||||
| M/F | 23/7 | Frequency/Duration | 1 h/session, 10 sessions, 2w | |||||||
| Age (y) | 35 ± 8 | |||||||||
| Furuya. et al., 201435 Germany | Pre-post | Type | Musician’s dystonia | Treatment | Behavioral training + tDCS | NA | Key stroke (performance) | Significant improvement in music performance | III | |
| N | 10 | Details | Re-training of piano under regular tempo during tDCS (primary motor cortex) | |||||||
| M/F | 6/4 | |||||||||
| Age (y) | 24–61 | Frequency/Duration | 24 min/ session, 5 sessions | |||||||
| Buttkus. et al., 201136 Germany | RCT cross-over | Type | Musician’s dystonia | Treatment | Sensorimotor rehab + tDCS | Sensorimotor rehab + sham tDCS | MIDI-based scale (severity) | No significant improvement in all three conditions | II | |
| N | 9 | Details | Retraining on piano + anodal or cathodal tDCS (2 mA, M1) | |||||||
| M/F | 9/0 | Frequency/Duration | 20 min, single session | |||||||
| Age (y) | 44 ± 11 | |||||||||
| Buttkus. et al., 201037 Germany | Case report, cross-over | Type | Musician’s dystonia | Treatment | Slow down exercise + anodal or cathodal tDCS | Sham tDCS | MIDI-based scale (severity) | Dystonia severity improved in all three conditions cathodal tDCS showed greater improvement | IV | |
| N | 1 | Details | Retraining on piano + anodal or cathodal tDCS (2 mA, M1) | |||||||
| M/F | M | Frequency/Duration | 20 min/session, 5 sessions/condition, 3 conditions, 21 w | |||||||
| Age (y) | 43 | |||||||||
| Functional Dystonia | ||||||||||
| Giorgi. et al., 202439 Italy | Case report | N | 1 | Treatment | Multimodal care | NA | Overall Symptoms (subjective) | Symptoms abated | IV | |
| M/F | M | Details | Mézières-Bertelè method + Tai chi + EMG biofeedback | |||||||
| Age (y) | 24 | Frequency/Duration | 1–3 sessions/w, 3m | |||||||
| Antelmi. et al., 202042 Italy | Case series | N | 2 | Treatment | Multimodal care | NA | Overall Symptoms (subjective) | One patient responded to rehabilitation One patient responded to BoNT | IV | |
| M/F | 0/2 | Details | BoNT, psychological and physical rehabilitation | |||||||
| Age (y) | 40, 65 | Frequency/Duration | NA | |||||||
| Vizcarra. et al., 201938 USA | RCT | N | 7 | 7 | Treatment | BoNT + CBT | Placebo BoNT + CBT | Psychiatric movement disorders rating scale (severity) | Both groups significantly improved dystonia severity No significant difference between groups | II |
| M/F | 1/6 | 2/4 | Details | Personalized CBT | ||||||
| Age (y) | 44 ± 15 | 53 ± 8 | Frequency/Duration | BoNT: 1 session + CBT: 1 session/w for 0–12 w, 12 w | ||||||
| Lee. et al., 201341 Germany | Case report | N | 1 | Treatment | Multimodal care | NA | Overall Symptoms (subjective) | No improvement | IV | |
| M/F | NA | Details | Splinting of the hand, PT, acupuncture | |||||||
| Age (y) | 21 | Frequency/Duration | several times, several days | |||||||
| Majumdar. et al., 200940 UK | Case series | N | 4 | Treatment | Multimodal care | NA | Overall Symptoms (subjective) | 2/4 patients with fixed dystonia responded | IV | |
| M/F | 0/4 | Details | BoNT, tenotomy, intensive PT, psychotherapy | |||||||
| Age (y) | 15 (13 −19) | Frequency/Duration | NA | |||||||
| Ziegler. et al., 200844 (Germany) | Case report | N | 1 | Treatment | Multimodal care | NA | Overall Symptoms (subjective) | Symptoms abated | IV | |
| M/F | F | Details | CBT + PT | |||||||
| Age (y) | 11 | Frequency/Duration | 1.5 y of inpatient & outpatient care | |||||||
Values are shown as mean (standard deviation or SD), if SD was not available in the article min - max were shown if possible. AAN class, American Academy of Neurology classification framework; number of patients, n; male, M; female, F; year, y; month, week, w; hour, h; minute, min; second, s; not applicable, NA; Randomized controlled trial, RCT; transcranial direct current stimulation, tDCS; Toronto Western spasmodic torticollis rating scale, TWSTRS; botulinum toxin injections, BoNT; Craniocervical dystonia questionnaire, CDQ4; Quality of Life, QOL; Physical therapy, PT; repetitive Transcranial Magnetic Stimulation, rTMS; resting motor threshold, RMT; primary motor cortex; PMC, Writer’s Cramp Rating Scale, WCRS; Physical therapy, PT; Range of Motion, ROM; 36-Item Short-Form Health Survey, SF-36; functional electrical stimulation, FES; Activities of Daily Living, ADL; Transcutaneous electrical nerve stimulation, TENS; sensory-motor retraining, SMR; musical instrument digital interface, MIDI; Occupational Therapy, OT; electromyography, EMG; adductor spasmodic dysphonia, ADSD; cognitive behavioral therapy, CBT.