J Neurol DOI 10.1007/s00415-013-7220-8
REVIEW
The effectiveness of physiotherapy for cervical dystonia: a systematic literature review Joke De Pauw • Kevin Van der Velden • Jill Meirte • Ulrike Van Daele • Steven Truijen • Patrick Cras • Rudy Mercelis • Willem De Hertogh
Received: 18 November 2013 / Accepted: 13 December 2013 Ó Springer-Verlag Berlin Heidelberg 2014
Abstract Cervical dystonia is a form of adult-onset, focal dystonia characterized by involuntary contractions of the neck muscles, leading to a disabling, abnormal head posture. CD has a great impact on the activities of daily living (ADL) and quality of life. Currently, the most widely used and recommended first line treatment is botulinum toxin type A (BoNT/A) injections. Physiotherapy is a potentially useful adjuvant, but little is known about its effectiveness. Consequently, our objective was to investigate the effectiveness of physiotherapy alone or as an adjuvant treatment to BoNT/A injections in cervical dystonia (CD) by means of a systematic literature review. Two online databases, PubMed and Web of Science, were searched for articles describing the effectiveness of physiotherapy treatment for CD. After screening, based on predefined in- and exclusion criteria, 16 studies were retained. Their methodological quality was assessed according to Cochrane guidelines. The methodological quality of most studies was low. Examples of shortcomings are small sample sizes, lack of randomization or blinding, and diversity in therapeutic techniques and outcome measures. Only seven studies were
J. De Pauw (&) K. Van der Velden J. Meirte U. Van Daele S. Truijen W. De Hertogh Department of Rehabilitation Sciences and Physiotherapy, Faculty of Medicine and Health Sciences, University of Antwerp, Antwerp, Belgium e-mail: [email protected] J. De Pauw Faculty of Medicine and Health Sciences, Campus Drie Eiken, Universiteitsplein 1, 2610 Wilrijk, Belgium P. Cras R. Mercelis Department of Neurology, University Hospital Antwerp, Faculty of Medicine and Health Sciences, Born Bunge Institute, University of Antwerp, Antwerp, Belgium
clinical trials; the remaining were either case reports or case series. The reported physiotherapy treatments included EMG biofeedback training, muscular elongation, postural exercises and electrotherapy. Improvements in head position, pain, cervical range of motion, quality of life and ADL have been reported, which is promising. Cautious interpretation on the effectiveness of physiotherapy as an adjuvant therapy is required. Before firm conclusions can be drawn, additional high quality trials are needed. Keywords Cervical dystonia Physiotherapy Treatment Botulinum toxin Quality of life (4)
Introduction Cervical dystonia (CD) is a neurological movement disorder consisting of sustained involuntary muscle contractions, which frequently result in abnormal movements or postures. Cervical dystonia is considered a form of primary adultonset focal dystonia [1, 2]. The prevalence numbers of CD vary, ranging from 2.3/100,000 to 390/100,000 [3, 4]. This wide range can be explained by the application of various definitions, e.g. by including non-focal dystonia or generalized dystonia or by regional differences. The reported numbers are also believed to underestimate the true prevalence due to delayed diagnostics and because only diagnosed cases are included [4–7]. CD can be considered an important health problem with a devastating impact on all levels of functioning and personal factors of individuals. For instance, CD affects a person’s working capacity and sleeping quality [8–12]. Also, more frequent psychiatric comorbidity as well as a higher prevalence of depression and anxiety disorders have been demonstrated in patients with CD in comparison with healthy controls [13–15].
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A variety of symptoms are reported by patients with idiopathic cervical dystonia. The main characteristics of cervical dystonia are: (1) uncontrolled contractions of neck muscles, resulting in postural deviations of the head and cervical spine. These contractions can be intermittent or sustained [1, 5]. They are repetitive and predictable as the contractions follow the same pattern [16, 17]. (2) Pain, described by most patients as a diffuse pain located in the neck and shoulder region. The pain can be intermittent or continuously present [5]. (3) Stiffness of the neck [17]. Some patients use a sensory trick or geste antagoniste to alleviate prolonged muscle contraction and postural deviation. The working mechanism of these tricks remains unclear [5, 18, 19]. In the course of cervical dystonia, many patients report deteriorating symptoms in the first 5 years of the disease, followed by a stabilization of symptoms [1, 5, 17]. The health related quality of life (HR-QOL) in patients diagnosed with CD is significantly reduced. For instance, lower scores on all domains of the SF-36 are demonstrated in CD compared to a general population [13, 20–23]. Female gender, lower level of education, the presence of pain, lower physical activity level, treatment satisfaction, working status, severity of CD and depression may influence these lower HR-QOL values [20, 21, 24]. The first-line treatment of cervical dystonia consists of botulinum toxin type A injections (BoNT/A) into the involved muscles in order to alleviate the symptoms [7]. Pallidal deep brain stimulations or selective denervation are secondary treatment options [7, 18]. Additionally, physiotherapy can be combined with BoNT/A injections [7, 25]. However, the role and effectiveness of physiotherapy in the treatment of CD remain unclear [25]. The systematic review of Delnooz et al. [25] reported a variety of paramedical treatments with varying outcome parameters, not taking into account the first-line treatment of BoNT/A injections. However, additional and more recent clinical trials reporting on the effect of physiotherapy in CD have been published. The objective of our systematic literature search is to review the effectiveness of physiotherapy alone or in addition to BoNT/A injections in patients with idiopathic cervical dystonia.
Methods This systematic review is reported following the PRISMA guidelines (preferred reporting items for systematic reviews and meta-analyses) [26]. Eligibility criteria In the search of relevant literature, studies had to report the outcome (O) of physiotherapy treatment (I) in patients with
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Table 1 Search strategy Keywords Cervical dystonia
AND
Physiotherapy
OR
‘‘Physical Therapy’’
Spasmodic torticollis
Rehabilitation ‘‘Paramedical treatment’’ ‘‘Sensory stimulation’’ Stretching Manipulation ‘‘Relaxation therapy’’ Biofeedback ‘‘Neuromotor rehabilitation’’ Training ‘‘Exercise therapy’’
idiopathic cervical dystonia (P). The study design (S) or comparison (C) was not specified in order to obtain all relevant and available studies concerning the effectiveness of physiotherapy in cervical dystonia patients. Information sources Pubmed and Web of Science were searched through May 2013 to identify relevant articles. Both databases were searched by using the following keywords: cervical dystonia or spasmodic torticollis combined with keywords related to physiotherapy such as rehabilitation, physical therapy, biofeedback, stretching, relaxation, paramedical treatment, sensory stimulation, stretching, manipulation, relaxation therapy, biofeedback, neuromotor rehabilitation, training, exercise therapy. The search strategy is presented in Table 1. Study selection To obtain relevant articles, the search results were screened using the following inclusion criteria: (1) the study is published after January 1990, (2) study subjects are adults and have cervical dystonia, (3) study subjects have no additional disorders of the neck, (4) articles are published in English, French, German or Dutch, (5) the physiotherapy intervention is clearly described. Screening for eligibility was first performed on title and abstract. Then, full-text versions were obtained. In the second phase, the full-text version was screened. Data items and collection Before data were extracted, a data extraction form was constructed. Two reviewers (JDP, MSc in rehabilitation sciences, pre-doctoral student and KVDV, MSc in rehabilitation sciences) independently extracted information
J Neurol Table 2 GRADE approach on interpreting methodological quality (43) Underlying methodology
Quality rating
Randomized trials; or double-upgraded observational studies
High
Downgraded randomized trials; or upgraded observational studies
Moderate
Double-downgraded randomized trials; or observational studies
Low
Triple-downgraded randomized trials; or downgraded observational studies; or case series/case reports
Very low
from the articles. Relevant information concerning (1) study type, (2) information regarding cervical dystonia: duration of the symptoms and severity of the symptoms, (3) type of intervention: used modalities, frequency and duration of the treatment, (4) follow-up, (5) medication intake or BoNT/A injections, (6) outcome parameters: Toronto Western Spasmodic Rating Scale (TWSTRS), Tsui scale, pain, SF36, QoL, Beck depression inventory were recorded.
Fig. 1 Flow chart of study selection
stimulation. The number of interventions ranged from one to 54 sessions. The primary outcome parameters reported were QoL, pain, severity of CD, depression, neck mobility, TWSTRS score and EMG activity of the sternocleidomastoid muscles. In the three most recent studies, the physiotherapy program was applied in addition to BoNT/A injections. Based on the extraction form, the characteristics for which data were extracted are presented in Table 3.
Risk of bias of individual studies Risk of bias and level of evidence Both reviewers independently assessed methodological quality according to the study design. The GRADE approach, as recommended by the Cochrane Collaboration was used to identify the level of evidence [27]. See Table 2.
Results Study selection The initial search provided a total of 1,208 studies (see Fig. 1: flow chart of the study selection). Titles and abstracts were screened based on the inclusion criteria. Based on this screening, 1,099 studies were excluded, meaning that 109 studies were withdrawn. After removing duplicates, 15 studies were included. One study was added based on the references of the treatment guidelines of the Dutch DystonieNET [28], resulting in 16 articles for data analysis. Study characteristics Of the 16 studies selected for this review, six were clinical trials, four case series and six case reports. The treatment of the CD patients consisted of electromyographic (EMG) biofeedback, multimodal physiotherapy programs, relaxation therapy, vibration and transcutaneous electrical nerve
Risk of bias and the level of evidence of the included studies are reported in Table 3. The inter-rater agreement was good (jw = 0.834, p \ 0.001). For the six clinical trials, methodological quality varied from low to high quality according to the GRADE approach. The methodological quality of the six case series and three case reports was very low. Most clinical trials did not randomize the study population or did not blind the patient or observer for the group allocation. No follow-up period was included in two clinical trials. Considering the low methodological quality of the case reports, they were only included for their more detailed description of the treatment content. Content of physiotherapy treatment The physiotherapy treatment of patients with cervical dystonia consisted of different types of physiotherapy modalities such as EMG biofeedback training, electrotherapy, relaxation training, active exercises to enhance muscular strength and posture, massage, muscular elongation techniques, mobilization techniques, manipulation techniques of the cervical spine, vibration, vestibular stimulation and cognitive behavioral therapy. EMG biofeedback training was applied in five studies [29–33]; only two studies reached a moderate methodological quality [30, 32]. The total number of participants in
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Cross over design
Clinical trial
Smania et al. [31]
Case report
Karnath et al. 2000
RCT
Duddy et al. [33]
Case series
Leis et al. 1992
Case report
Spencer et al. [34]
RCT
Jahanshabi et al. [30]
Case series
3F 41.7y
N=4
54y
N=1
N = 11
31–65y
4F
N = 11
m
N=1
52,7y
7F
N = 12
42–61y
1–3.8y
6y
9y
/
0.7 y
6.5y
/
/
/
/
/
/
/
/
N = 10
Leplow et al. [29]
4F
Severity of CD
Duration of CD
Patients
Author study design
Table 3 Overview of included articles
No
No
/
Medication
/
Medication
/
Med BoNT-A
6 weeks of treatment at ratio of 59/ week
1 h sessions of physiotherapy program consisting of 20 min elongation techniques and 40 min of active postural reeducation
15 min of muscle vibration on the right splenius in 2 treatment session spread over 3 months
Television monitored relaxation combined with EMG biofeedback
1 session
Electrostimulation (TENS), Vibration
Relaxation training, exercises to strengthen the antagonists, correction of the head position with mirror 20 weeks of treatment: 7 supervised sessions
27 sessions at ratio of 1 or 29/week
EMG biofeedback training
14 sessions
EMG biofeedback training, relaxation, stress-management and advice
Intervention
EMG biofeedback
15 min of muscle vibration on healthy individuals
Television monitored relaxation
/
/
Relaxation and graded practice to improve head position and movement
/
Control
9m
/
/
/
24 m
3m
/
FU
Increased functioning in ADL remained during 9 m follow-up
Both interventions improved pain and head-trunk alignment
Note that the sensory stimulus of the electrodes alone showed an improvement in head position
Decreased lateral flexion and tremor during several minutes after vibration
There is no indication that biofeedback has an additional benefit to relaxation therapy
Note that only 5 of the 11 cases showed improvements
Decreased involuntary movements and decreased muscle activity
The increase in ROM and decrease in EMG activity of the SMC after treatment continued after 2-year follow-up
Increased ROM, improved head position, decreased muscle tension in SCM
Both groups showed improvement for depression and body concept
Head position and neck mobility was improved in both groups after treatment and follow-up
EMG in rest was larger in the biofeedback group
Large variability between patients responding to biofeedback
No decreased tonus of SCM through the sessions
Reduced tonus of SCM during biofeedback
Results
Low
Very low
Low
Very low
Very low
Moderate
Very low
GRADE
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Case series
Zetterberg et al. [36]
Case report
Rosengren et al. 2006
Case report
Faircloth et al. 2006
Case report
Ramdharry [35]
RCT
Tassorelli et al. [32]
Case report
N=1
Kukurin et al. 2004
(34-62)
(5–29y)
(30–59y)
48.5y
TWSTRS 49
12.3y
/
/
/
N=6
12y
10y
9y
/
Grade 16 on CDSS
/
11.5y ± 2.9y
Severity of CD
Duration of CD
4F
61y
N=1 F
36y
M
N=1
M
N=1
51.3y ± 15.6y
13F
N = 40
38y
M
Patients
Author study design
Table 3 continued
No
No
No
BoNT-A
BoNT-A
/
Med BoNT-A
4 week program with 36 supervised treatments, 2 sessions of 45 min per day (with exception of Fridays)
Relaxation, isometric and dynamic exercises to strengthen antagonists, training of balance, coordination and body perception
1 session for left vestibular system, 1 for the right
Galvanic and acoustic vestibular stimulation
Cognitive-behavioral therapy
14 sessions at ratio of one session in 4 to 6 weeks ? home exercises daily for 1y
Physiotherapy program to strengthen the antagonist muscles of the dystonia, massage and core stability training
10 treatment sessions of 60 to 90 min during 2 weeks at ratio of 59/week
BoNT-A combined with physiotherapy: massage, stretching, postural reeducation, strengthening of the axial muscles and 30 min biofeedback
54 sessions during 7 months at ratio of 3 to 59/week
Manipulation techniques of the cervical and thoracic spine
Intervention
/
/
/
/
BoNT-A
/
Control
6m
/
6m
/
/
/
FU
The improvement of the quality of life remained during the 6 m follow-up period.
Significant improvement of pain, score on TWSTR, disability and quality of life
Self reported improvement of ROM, head position and pain
Acoustic stimulation decreases EMG activity of ipsilateral SCM
Effect remains after 6 m follow-up
Increased level of activity and participation in social life
Better coping mechanism, less anxiety, self reported improvement of pain and head position
Longer time period in between BoNT-A injection, requiring lower dose
Decreased scores on all subscales of TWSTR, decreased absenteeism
The effects of BoNT-A lasted sig. longer and a sig. lower dose was needed for the next injection
No sig. decrease of severity of dystonia in both groups on Tsui scale and TWSTRS Sig less pain and increased ADL functioning in physiotherapy group
Severity of dystonia decreased on the CDSS from 16 to 5
Results
Low
Very low
Very low
Very low
Moderate
Very low
GRADE
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57.8y ± 7.8y
14 F
N = 20
51.3y ± 13.6y
N = 20 20F
32.5 y ± 4.1y
10.2y ± 7.9y 37.7 ± 10.7 C: 34.7 ± 13.4
I:
TWSTRS
C: 34.6
TWSTRS I: 35,7
/
/
I: 9y C: 16y
Severity of CD
Duration of CD
BoNT-A N=7
No med N = 13
BoNT-A
BoNT-A
Med BoNT-A
12-week program with 8 supervised sessions (30 min) at ratio of 19/ week and daily home exercises
Combined with BoNT/A N = 3
Relaxation ? individualized exercise program of active exercises to increase muscle strength of antagonist muscles and induce normal head position
4-week program of 20 sessions at ratio of 59/week
BoNT-A combined with 1 h physiotherapy sessions: motor learning exercises, stretching, active and passive mobilization of the cervical spine, functional electrical stimulation of antagonist muscles
6 week program with 18 sessions at ratio of 39/week
BoNT-A combined with 1 h physiotherapy sessions consisting of stretching SCM, training of voluntary movements, TENS
Intervention
Combined with BoNT/ AN=4
Whole body Relaxation program
BoNT-A
BoNT-A combined with placebo TENS
Control
4m
no
/
FU
A power calculation showed the need to include 34 patients and an improvement of 7 points on the TWSTR for a positive treatment effect.
Patients were able to perform an active exercise program, but there was only a trend towards better scores on the TWSTR and decreased depression (Beck Depression Inventory)
Both physical and mental health sig improved (SF36) in physiotherapy group.
Physiotherapy group had also sig decrease in disability and pain subscale of TWSTR
Significant decrease of the severity of the dystonia in both groups
Decreased disability, significant decrease in rotational deviation of the head (38.7°[21.8°) and improved fine motor hand function
Results
High
Moderate
Moderate
GRADE
/ not reported, I intervention group, C control group, CDSS cervical dystonia severity scale, TWSTRS Toronto Western Spasmodic Torticollis Rating Scale, TENS transcutaneus electrical nerve stimulation, FU Follow-up reported in months (m), SCM sternocleidomastoid muscle
Patients: N, number of participants; m, male; f, female, age in years (y), Ilness duration in years (y), Medication BTX: did participants receive botulinum toxin injections during the study?
RCT
Boyce et al. [39]
RCT
Queiroz et al. [38]
RCT
N = 40
ElBahrawy et al. [37]
15F
Patients
Author study design
Table 3 continued
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the five studies was 66. EMG biofeedback training is equally effective as a combination of relaxation and exercises in reducing the tonus of the sternocleidomastoid muscle, improving the cervical range of motion (ROM) and position of the head. Both interventions improve body concept and reduce depression [30]. In an uncontrolled trial in four patients, EMG biofeedback training alone seemed to reduce pain intensity, improved head-trunk alignment and increased ADL functioning in patients in the first years of cervical dystonia (1–3,8 years duration of the pathology) [31]. The effect of the EMG biofeedback training has large inter-individual variability where some individuals experience no effect at all [30, 33]. There is limited evidence that EMG biofeedback training alone is effective in the treatment of cervical dystonia. A multimodal physiotherapy program was applied in nine studies [30–32, 34–39], one study of high and four of moderate methodological quality. The total number of participants included in RCTs was 132. Multimodal physiotherapy programs combine multiple of the abovementioned modalities. Tassorelli et al. [32] conducted a RCT in 40 patients with CD. A 2-week physiotherapy program combined with BoNT/A and EMG biofeedback showed no beneficial effect on the severity of CD measured by the TWSTRS and Tsui scale, yet patients experienced significantly less pain, increased functioning and the effect of BoNT/A lasted significantly longer, requiring a lower dose for the following injection. In a case series of six patients [36], a 4-week physiotherapy program of relaxation, active exercises to strengthen the antagonist muscles, training of balance, coordination and body perception resulted in decreased pain and disability and an improved quality of life. The improved QoL remained after 6 months of follow-up. A 6-week program of stretching, active exercises and TENS in combination to BoNT/A led to decreased disability, a significant decrease of rotational deviation of the head and improved fine motor hand function [37]. In the RCT of Queiroz et al. [38], the control group was treated with BoNT/A and the intervention group was treated with BoNT/A combined with a physiotherapy program consisting of motor learning exercises, stretching, active and passive mobilization of the cervical spine and functional electrical stimulation of the antagonist muscles. After the 4-week program, both groups showed a significant decrease of the severity of CD on the TWSTRS, showing the positive results of the BoNT/A injection. Yet the patients who received the multimodal physiotherapy program showed also a significant decrease on the disability and pain subscale of the TWSTRS. Secondly, both physical and mental health on the SF-36 significantly improved.
In the recent RCT of Boyce et al. [39], the intervention group received whole body relaxation and active exercises to increase the muscle strength of the antagonist muscles to induce a normal head position. This was compared to the control group who received only whole body relaxation. Patients treated with BoNT/A as well as patients receiving no other treatment were included. After a period of 12 weeks with eight treatment sessions, no positive effect of active exercises was found; only a trend towards better scores on the TWSTRS and lower depression were observed. A multimodal physiotherapy program adjunct to BoNT/ A injections may improve head position, decrease pain levels in patients with CD and improve functioning in every day activities on short term.
Discussion The aim of this systematic literature study was to evaluate the effectiveness of physiotherapy alone or in addition to BoNT/A injections in the treatment of CD. We identified 16 studies reporting the effectiveness of physiotherapy in CD patients. Only six RCTs were included, one clinical trial with a cross-over design, three case series and six case reports. The methodological quality of the RCTs was moderate to high with a high risk of selection bias due to a lack of blinding of both patients and observers as well as randomization. And in most of the studies, no follow-up period was reported. EMG biofeedback was applied in five studies. There is limited, low-quality evidence for the application of EMG biofeedback training as a solitary treatment for CD. Three trials of moderate methodological quality evaluated the effect of physiotherapy as an adjuvant treatment to BoNT/A injections [32, 37, 38]. Based on their findings, we can conclude that a multimodal physiotherapy treatment adjunct to BoNT/A injections may improve head position, decrease pain levels in patients with CD and improve functioning in every day activities on short term. One trial of high methodological quality showed a trend to improved scores on the TWSTRS [39]. The lack of significance might be a result of similarities in both treatment groups. Whole body relaxation was applied in both treatment groups. In the active group, it was combined with an active exercise program. The control group received no additional treatment. Given the fact that mental and physical load may aggravate the symptoms of CD, it is possible that whole body relaxation in itself has a beneficial effect on the symptoms of CD. The content of multimodal physiotherapy treatment varied, including active exercises, muscular elongation techniques or stretching, massage, relaxation, active and
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passive mobilization of the cervical spine and electrotherapy such as EMG biofeedback, TENS, or electrical stimulation of the antagonist muscles. Most active exercises are based on the therapy program of Bleton [40], in which muscles with the opposite function of the direction in which dystonic muscles pull the head are strengthened. These types of active exercises are included in multimodal therapy programs as well as other postural exercises aiming to correct the impaired position of the head. Regardless of the type of exercises performed, there is some evidence that an active approach can ameliorate the symptoms of cervical dystonia. The number of treatments also varied widely and ranged from eight to 36. Consequently, it is currently not clear how many treatment sessions should be prescribed. In the description of the patient population, gender, age and illness duration were most frequently reported. Mean age varied from 32.5 to 57.8 years and the mean illness duration varied from 1.8 to 16 years, which may indicate that patients who are recently diagnosed as well as patients with a long history of CD might benefit from an additional multimodal physiotherapy treatment. However, it would also be interesting to know whether the severity of CD plays a role in the treatment effect or if the effect of a multimodal physiotherapy program is depended on the severity of CD. Only three studies reported the severity of CD measured by the TWSTRS with a mean score varying from 34.6 to 49 [36, 38, 39]. We suggest the use of a severity scale such as the TWSTRS or CDIP-58 when describing future patient samples. This is in line with the recommendations by Albanese et al. [41]. This review has strengths and limitations. We focused on the treatment of cervical dystonia solely and two reviewers independently assessed the methodological quality of the included articles in order to reduce the risk of bias. We were able to select recently published RCTs [37– 39], which provided more information of controlled intervention physiotherapy treatments consisting of active exercises, stretching and biofeedback or transcutaneous electrical stimulation especially in combination with BoTN/A injections. In this systematic literature search, we only searched two databases and only in English, German, Dutch or French. It is, therefore, possible that we have missed some clinical studies. We would like to stipulate that the findings should be interpreted with caution. The limited number of RCTs, the limited methodological quality and the total of 132 patients reported in the RCTs diminish the power of recommendations that can be made. The rather low prevalence of idiopathic cervical dystonia [3, 4] may be the reason for the low number of participants. The content of the multimodal
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physiotherapy interventions and active exercises differed widely among the studies, making it difficult to identify the most effective component. Further research should address randomization, doubleblinding and should consider working multicentrical to enlarge the study population. Also, more objective parameters concerning the patient population should be reported. For instance, the diagnostic criteria for CD should be reported along with objective parameters such as the Cervical Dystonia Impact Profile and the Toronto Western Spasmodic Torticollis Rating Scale. This can be used to describe included samples and therapy progress in a more uniform manner.
Conclusions Regardless of the central neurological etiology of cervical dystonia, the evidence that a multimodal physiotherapy program consisting of active exercises, stretching and relaxation in addition to botulinum toxin treatment induces beneficial effects on the pain and disability of patients with cervical dystonia on short term is growing. Additional high-quality trials are needed before firm recommendations can be made. Acknowledgments We would like to thank Birgit Daalderop for helping with the data collection. Funding
University of Antwerp (BDHG/3449/18.12.202).
Conflicts of interest On behalf of all authors, the corresponding author states that there is no conflict of interest.
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25. Delnooz CCS, Horstink MWIM, Tijssen MA, van de Warrenburg BPC (2009) Paramedical treatment in primary dystonia: a systematic review. Mov Disord 24:2187–2198. doi:10.1002/mds. 22608 26. Liberati A, Altman DG, Tetzlaff J et al (2009) The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: explanation and elaboration. PLoS Med 6:e1000100. doi:10.1371/journal.pmed. 1000100 27. Schu¨nermann HJ, Oxman AD, Vist GE, Higgins JPT, Deeks JJ, Glasziou GG (2011) Chapter 12: Interpreting results and drawing conclusions. Cochrane Handb. Syst. Rev. Interv. Version 5.1.0 28. Postma M, Zoons E, Speelman J, de Koning-Tijssen M (2012) (Botuline) Behandel richtlijn voor Cervicale dystonie. 1–90 29. Leplow B (1990) Heterogenity of biofeedback training effects in spasmodic torticollis: a single-case approach. Behav Res Ther 4:359–365 30. Jahanshahi M, Sartory G, Marsden CD (1991) EMG biofeedback treatment of torticollis: a controlled outcome study. Biofeedback Self Regul 16:413–448 31. Smania N, Corato E, Tinazzi M et al (2003) The effect of two different rehabilitation treatments in cervical dystonia: preliminary results in four patients. Funct Neurol 18:219–225 32. Tassorelli C, Mancini F, Balloni L et al (2006) Botulinum toxin and neuromotor rehabilitation: an integrated approach to idiopathic cervical dystonia. Mov Disord 21:2240–2243. doi:10. 1002/mds.21145 33. Duddy J, McLellan D (1995) Lack of influence of EMG biofeedback in relaxation training for spasmodic torticollis. Clin Rehabil 9:297–303. doi:10.1177/026921559500900404 34. John Spencer, Goetsch Virginia L, Brugnoli Robert J, Herman S (1991) Behavior therapy for spasmodic torticollis: a case study suggesting a causal role for anxiety. J Behav Ther Exp Psychiatry 22:305–311 35. Ramdharry G (2006) Case report: physiotherapy cuts the dose of botulinum toxin. Physiother Res Int 11:117–122. doi:10.1002/pri 36. Zetterberg L, Halvorsen K, Fa¨rnstrand C et al (2008) Physiotherapy in cervical dystonia: six experimental single-case studies. Physiother Theory Pract 24:275–290. doi:10.1080/0959398070 1884816 37. El-bahrawy MN, El-tamawy MS, Shalaby NM, Abdel-alim AM (2009) Cervical Dystonia : abnormal head posture and its relation to hand function. Egypt J Neurol Psychiatry Neurosurg 46:203–208 38. Queiroz MAR, Chien HF, Sekeff-sallem FA, Barbosa ER (2012) Physical therapy program for cervical dystonia: a study of 20 cases. Funct Neurol 27:187–192 39. Boyce MJ, Canning CG, Mahant N et al (2013) Active exercise for individuals with cervical dystonia: a pilot randomized controlled trial. Clin Rehabil 27:226–235. doi:10.1177/02692155 12456221 40. Bleton JP (1994) Spasmodic torticollis. In: Handbook of rehabilitative physiotherapy. Editions Frison-Roche, Paris 41. Albanese A, Del Sorbo F, Comella C et al (2013) Dystonia rating scales: critique and recommendations. Mov Disord 28:874–883. doi:10.1002/mds.25579
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REVIEW
The effectiveness of physiotherapy for cervical dystonia: a systematic literature review Joke De Pauw • Kevin Van der Velden • Jill Meirte • Ulrike Van Daele • Steven Truijen • Patrick Cras • Rudy Mercelis • Willem De Hertogh
Received: 18 November 2013 / Accepted: 13 December 2013 Ó Springer-Verlag Berlin Heidelberg 2014
Abstract Cervical dystonia is a form of adult-onset, focal dystonia characterized by involuntary contractions of the neck muscles, leading to a disabling, abnormal head posture. CD has a great impact on the activities of daily living (ADL) and quality of life. Currently, the most widely used and recommended first line treatment is botulinum toxin type A (BoNT/A) injections. Physiotherapy is a potentially useful adjuvant, but little is known about its effectiveness. Consequently, our objective was to investigate the effectiveness of physiotherapy alone or as an adjuvant treatment to BoNT/A injections in cervical dystonia (CD) by means of a systematic literature review. Two online databases, PubMed and Web of Science, were searched for articles describing the effectiveness of physiotherapy treatment for CD. After screening, based on predefined in- and exclusion criteria, 16 studies were retained. Their methodological quality was assessed according to Cochrane guidelines. The methodological quality of most studies was low. Examples of shortcomings are small sample sizes, lack of randomization or blinding, and diversity in therapeutic techniques and outcome measures. Only seven studies were
J. De Pauw (&) K. Van der Velden J. Meirte U. Van Daele S. Truijen W. De Hertogh Department of Rehabilitation Sciences and Physiotherapy, Faculty of Medicine and Health Sciences, University of Antwerp, Antwerp, Belgium e-mail: [email protected] J. De Pauw Faculty of Medicine and Health Sciences, Campus Drie Eiken, Universiteitsplein 1, 2610 Wilrijk, Belgium P. Cras R. Mercelis Department of Neurology, University Hospital Antwerp, Faculty of Medicine and Health Sciences, Born Bunge Institute, University of Antwerp, Antwerp, Belgium
clinical trials; the remaining were either case reports or case series. The reported physiotherapy treatments included EMG biofeedback training, muscular elongation, postural exercises and electrotherapy. Improvements in head position, pain, cervical range of motion, quality of life and ADL have been reported, which is promising. Cautious interpretation on the effectiveness of physiotherapy as an adjuvant therapy is required. Before firm conclusions can be drawn, additional high quality trials are needed. Keywords Cervical dystonia Physiotherapy Treatment Botulinum toxin Quality of life (4)
Introduction Cervical dystonia (CD) is a neurological movement disorder consisting of sustained involuntary muscle contractions, which frequently result in abnormal movements or postures. Cervical dystonia is considered a form of primary adultonset focal dystonia [1, 2]. The prevalence numbers of CD vary, ranging from 2.3/100,000 to 390/100,000 [3, 4]. This wide range can be explained by the application of various definitions, e.g. by including non-focal dystonia or generalized dystonia or by regional differences. The reported numbers are also believed to underestimate the true prevalence due to delayed diagnostics and because only diagnosed cases are included [4–7]. CD can be considered an important health problem with a devastating impact on all levels of functioning and personal factors of individuals. For instance, CD affects a person’s working capacity and sleeping quality [8–12]. Also, more frequent psychiatric comorbidity as well as a higher prevalence of depression and anxiety disorders have been demonstrated in patients with CD in comparison with healthy controls [13–15].
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A variety of symptoms are reported by patients with idiopathic cervical dystonia. The main characteristics of cervical dystonia are: (1) uncontrolled contractions of neck muscles, resulting in postural deviations of the head and cervical spine. These contractions can be intermittent or sustained [1, 5]. They are repetitive and predictable as the contractions follow the same pattern [16, 17]. (2) Pain, described by most patients as a diffuse pain located in the neck and shoulder region. The pain can be intermittent or continuously present [5]. (3) Stiffness of the neck [17]. Some patients use a sensory trick or geste antagoniste to alleviate prolonged muscle contraction and postural deviation. The working mechanism of these tricks remains unclear [5, 18, 19]. In the course of cervical dystonia, many patients report deteriorating symptoms in the first 5 years of the disease, followed by a stabilization of symptoms [1, 5, 17]. The health related quality of life (HR-QOL) in patients diagnosed with CD is significantly reduced. For instance, lower scores on all domains of the SF-36 are demonstrated in CD compared to a general population [13, 20–23]. Female gender, lower level of education, the presence of pain, lower physical activity level, treatment satisfaction, working status, severity of CD and depression may influence these lower HR-QOL values [20, 21, 24]. The first-line treatment of cervical dystonia consists of botulinum toxin type A injections (BoNT/A) into the involved muscles in order to alleviate the symptoms [7]. Pallidal deep brain stimulations or selective denervation are secondary treatment options [7, 18]. Additionally, physiotherapy can be combined with BoNT/A injections [7, 25]. However, the role and effectiveness of physiotherapy in the treatment of CD remain unclear [25]. The systematic review of Delnooz et al. [25] reported a variety of paramedical treatments with varying outcome parameters, not taking into account the first-line treatment of BoNT/A injections. However, additional and more recent clinical trials reporting on the effect of physiotherapy in CD have been published. The objective of our systematic literature search is to review the effectiveness of physiotherapy alone or in addition to BoNT/A injections in patients with idiopathic cervical dystonia.
Methods This systematic review is reported following the PRISMA guidelines (preferred reporting items for systematic reviews and meta-analyses) [26]. Eligibility criteria In the search of relevant literature, studies had to report the outcome (O) of physiotherapy treatment (I) in patients with
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Table 1 Search strategy Keywords Cervical dystonia
AND
Physiotherapy
OR
‘‘Physical Therapy’’
Spasmodic torticollis
Rehabilitation ‘‘Paramedical treatment’’ ‘‘Sensory stimulation’’ Stretching Manipulation ‘‘Relaxation therapy’’ Biofeedback ‘‘Neuromotor rehabilitation’’ Training ‘‘Exercise therapy’’
idiopathic cervical dystonia (P). The study design (S) or comparison (C) was not specified in order to obtain all relevant and available studies concerning the effectiveness of physiotherapy in cervical dystonia patients. Information sources Pubmed and Web of Science were searched through May 2013 to identify relevant articles. Both databases were searched by using the following keywords: cervical dystonia or spasmodic torticollis combined with keywords related to physiotherapy such as rehabilitation, physical therapy, biofeedback, stretching, relaxation, paramedical treatment, sensory stimulation, stretching, manipulation, relaxation therapy, biofeedback, neuromotor rehabilitation, training, exercise therapy. The search strategy is presented in Table 1. Study selection To obtain relevant articles, the search results were screened using the following inclusion criteria: (1) the study is published after January 1990, (2) study subjects are adults and have cervical dystonia, (3) study subjects have no additional disorders of the neck, (4) articles are published in English, French, German or Dutch, (5) the physiotherapy intervention is clearly described. Screening for eligibility was first performed on title and abstract. Then, full-text versions were obtained. In the second phase, the full-text version was screened. Data items and collection Before data were extracted, a data extraction form was constructed. Two reviewers (JDP, MSc in rehabilitation sciences, pre-doctoral student and KVDV, MSc in rehabilitation sciences) independently extracted information
J Neurol Table 2 GRADE approach on interpreting methodological quality (43) Underlying methodology
Quality rating
Randomized trials; or double-upgraded observational studies
High
Downgraded randomized trials; or upgraded observational studies
Moderate
Double-downgraded randomized trials; or observational studies
Low
Triple-downgraded randomized trials; or downgraded observational studies; or case series/case reports
Very low
from the articles. Relevant information concerning (1) study type, (2) information regarding cervical dystonia: duration of the symptoms and severity of the symptoms, (3) type of intervention: used modalities, frequency and duration of the treatment, (4) follow-up, (5) medication intake or BoNT/A injections, (6) outcome parameters: Toronto Western Spasmodic Rating Scale (TWSTRS), Tsui scale, pain, SF36, QoL, Beck depression inventory were recorded.
Fig. 1 Flow chart of study selection
stimulation. The number of interventions ranged from one to 54 sessions. The primary outcome parameters reported were QoL, pain, severity of CD, depression, neck mobility, TWSTRS score and EMG activity of the sternocleidomastoid muscles. In the three most recent studies, the physiotherapy program was applied in addition to BoNT/A injections. Based on the extraction form, the characteristics for which data were extracted are presented in Table 3.
Risk of bias of individual studies Risk of bias and level of evidence Both reviewers independently assessed methodological quality according to the study design. The GRADE approach, as recommended by the Cochrane Collaboration was used to identify the level of evidence [27]. See Table 2.
Results Study selection The initial search provided a total of 1,208 studies (see Fig. 1: flow chart of the study selection). Titles and abstracts were screened based on the inclusion criteria. Based on this screening, 1,099 studies were excluded, meaning that 109 studies were withdrawn. After removing duplicates, 15 studies were included. One study was added based on the references of the treatment guidelines of the Dutch DystonieNET [28], resulting in 16 articles for data analysis. Study characteristics Of the 16 studies selected for this review, six were clinical trials, four case series and six case reports. The treatment of the CD patients consisted of electromyographic (EMG) biofeedback, multimodal physiotherapy programs, relaxation therapy, vibration and transcutaneous electrical nerve
Risk of bias and the level of evidence of the included studies are reported in Table 3. The inter-rater agreement was good (jw = 0.834, p \ 0.001). For the six clinical trials, methodological quality varied from low to high quality according to the GRADE approach. The methodological quality of the six case series and three case reports was very low. Most clinical trials did not randomize the study population or did not blind the patient or observer for the group allocation. No follow-up period was included in two clinical trials. Considering the low methodological quality of the case reports, they were only included for their more detailed description of the treatment content. Content of physiotherapy treatment The physiotherapy treatment of patients with cervical dystonia consisted of different types of physiotherapy modalities such as EMG biofeedback training, electrotherapy, relaxation training, active exercises to enhance muscular strength and posture, massage, muscular elongation techniques, mobilization techniques, manipulation techniques of the cervical spine, vibration, vestibular stimulation and cognitive behavioral therapy. EMG biofeedback training was applied in five studies [29–33]; only two studies reached a moderate methodological quality [30, 32]. The total number of participants in
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Cross over design
Clinical trial
Smania et al. [31]
Case report
Karnath et al. 2000
RCT
Duddy et al. [33]
Case series
Leis et al. 1992
Case report
Spencer et al. [34]
RCT
Jahanshabi et al. [30]
Case series
3F 41.7y
N=4
54y
N=1
N = 11
31–65y
4F
N = 11
m
N=1
52,7y
7F
N = 12
42–61y
1–3.8y
6y
9y
/
0.7 y
6.5y
/
/
/
/
/
/
/
/
N = 10
Leplow et al. [29]
4F
Severity of CD
Duration of CD
Patients
Author study design
Table 3 Overview of included articles
No
No
/
Medication
/
Medication
/
Med BoNT-A
6 weeks of treatment at ratio of 59/ week
1 h sessions of physiotherapy program consisting of 20 min elongation techniques and 40 min of active postural reeducation
15 min of muscle vibration on the right splenius in 2 treatment session spread over 3 months
Television monitored relaxation combined with EMG biofeedback
1 session
Electrostimulation (TENS), Vibration
Relaxation training, exercises to strengthen the antagonists, correction of the head position with mirror 20 weeks of treatment: 7 supervised sessions
27 sessions at ratio of 1 or 29/week
EMG biofeedback training
14 sessions
EMG biofeedback training, relaxation, stress-management and advice
Intervention
EMG biofeedback
15 min of muscle vibration on healthy individuals
Television monitored relaxation
/
/
Relaxation and graded practice to improve head position and movement
/
Control
9m
/
/
/
24 m
3m
/
FU
Increased functioning in ADL remained during 9 m follow-up
Both interventions improved pain and head-trunk alignment
Note that the sensory stimulus of the electrodes alone showed an improvement in head position
Decreased lateral flexion and tremor during several minutes after vibration
There is no indication that biofeedback has an additional benefit to relaxation therapy
Note that only 5 of the 11 cases showed improvements
Decreased involuntary movements and decreased muscle activity
The increase in ROM and decrease in EMG activity of the SMC after treatment continued after 2-year follow-up
Increased ROM, improved head position, decreased muscle tension in SCM
Both groups showed improvement for depression and body concept
Head position and neck mobility was improved in both groups after treatment and follow-up
EMG in rest was larger in the biofeedback group
Large variability between patients responding to biofeedback
No decreased tonus of SCM through the sessions
Reduced tonus of SCM during biofeedback
Results
Low
Very low
Low
Very low
Very low
Moderate
Very low
GRADE
J Neurol
Case series
Zetterberg et al. [36]
Case report
Rosengren et al. 2006
Case report
Faircloth et al. 2006
Case report
Ramdharry [35]
RCT
Tassorelli et al. [32]
Case report
N=1
Kukurin et al. 2004
(34-62)
(5–29y)
(30–59y)
48.5y
TWSTRS 49
12.3y
/
/
/
N=6
12y
10y
9y
/
Grade 16 on CDSS
/
11.5y ± 2.9y
Severity of CD
Duration of CD
4F
61y
N=1 F
36y
M
N=1
M
N=1
51.3y ± 15.6y
13F
N = 40
38y
M
Patients
Author study design
Table 3 continued
No
No
No
BoNT-A
BoNT-A
/
Med BoNT-A
4 week program with 36 supervised treatments, 2 sessions of 45 min per day (with exception of Fridays)
Relaxation, isometric and dynamic exercises to strengthen antagonists, training of balance, coordination and body perception
1 session for left vestibular system, 1 for the right
Galvanic and acoustic vestibular stimulation
Cognitive-behavioral therapy
14 sessions at ratio of one session in 4 to 6 weeks ? home exercises daily for 1y
Physiotherapy program to strengthen the antagonist muscles of the dystonia, massage and core stability training
10 treatment sessions of 60 to 90 min during 2 weeks at ratio of 59/week
BoNT-A combined with physiotherapy: massage, stretching, postural reeducation, strengthening of the axial muscles and 30 min biofeedback
54 sessions during 7 months at ratio of 3 to 59/week
Manipulation techniques of the cervical and thoracic spine
Intervention
/
/
/
/
BoNT-A
/
Control
6m
/
6m
/
/
/
FU
The improvement of the quality of life remained during the 6 m follow-up period.
Significant improvement of pain, score on TWSTR, disability and quality of life
Self reported improvement of ROM, head position and pain
Acoustic stimulation decreases EMG activity of ipsilateral SCM
Effect remains after 6 m follow-up
Increased level of activity and participation in social life
Better coping mechanism, less anxiety, self reported improvement of pain and head position
Longer time period in between BoNT-A injection, requiring lower dose
Decreased scores on all subscales of TWSTR, decreased absenteeism
The effects of BoNT-A lasted sig. longer and a sig. lower dose was needed for the next injection
No sig. decrease of severity of dystonia in both groups on Tsui scale and TWSTRS Sig less pain and increased ADL functioning in physiotherapy group
Severity of dystonia decreased on the CDSS from 16 to 5
Results
Low
Very low
Very low
Very low
Moderate
Very low
GRADE
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123
57.8y ± 7.8y
14 F
N = 20
51.3y ± 13.6y
N = 20 20F
32.5 y ± 4.1y
10.2y ± 7.9y 37.7 ± 10.7 C: 34.7 ± 13.4
I:
TWSTRS
C: 34.6
TWSTRS I: 35,7
/
/
I: 9y C: 16y
Severity of CD
Duration of CD
BoNT-A N=7
No med N = 13
BoNT-A
BoNT-A
Med BoNT-A
12-week program with 8 supervised sessions (30 min) at ratio of 19/ week and daily home exercises
Combined with BoNT/A N = 3
Relaxation ? individualized exercise program of active exercises to increase muscle strength of antagonist muscles and induce normal head position
4-week program of 20 sessions at ratio of 59/week
BoNT-A combined with 1 h physiotherapy sessions: motor learning exercises, stretching, active and passive mobilization of the cervical spine, functional electrical stimulation of antagonist muscles
6 week program with 18 sessions at ratio of 39/week
BoNT-A combined with 1 h physiotherapy sessions consisting of stretching SCM, training of voluntary movements, TENS
Intervention
Combined with BoNT/ AN=4
Whole body Relaxation program
BoNT-A
BoNT-A combined with placebo TENS
Control
4m
no
/
FU
A power calculation showed the need to include 34 patients and an improvement of 7 points on the TWSTR for a positive treatment effect.
Patients were able to perform an active exercise program, but there was only a trend towards better scores on the TWSTR and decreased depression (Beck Depression Inventory)
Both physical and mental health sig improved (SF36) in physiotherapy group.
Physiotherapy group had also sig decrease in disability and pain subscale of TWSTR
Significant decrease of the severity of the dystonia in both groups
Decreased disability, significant decrease in rotational deviation of the head (38.7°[21.8°) and improved fine motor hand function
Results
High
Moderate
Moderate
GRADE
/ not reported, I intervention group, C control group, CDSS cervical dystonia severity scale, TWSTRS Toronto Western Spasmodic Torticollis Rating Scale, TENS transcutaneus electrical nerve stimulation, FU Follow-up reported in months (m), SCM sternocleidomastoid muscle
Patients: N, number of participants; m, male; f, female, age in years (y), Ilness duration in years (y), Medication BTX: did participants receive botulinum toxin injections during the study?
RCT
Boyce et al. [39]
RCT
Queiroz et al. [38]
RCT
N = 40
ElBahrawy et al. [37]
15F
Patients
Author study design
Table 3 continued
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the five studies was 66. EMG biofeedback training is equally effective as a combination of relaxation and exercises in reducing the tonus of the sternocleidomastoid muscle, improving the cervical range of motion (ROM) and position of the head. Both interventions improve body concept and reduce depression [30]. In an uncontrolled trial in four patients, EMG biofeedback training alone seemed to reduce pain intensity, improved head-trunk alignment and increased ADL functioning in patients in the first years of cervical dystonia (1–3,8 years duration of the pathology) [31]. The effect of the EMG biofeedback training has large inter-individual variability where some individuals experience no effect at all [30, 33]. There is limited evidence that EMG biofeedback training alone is effective in the treatment of cervical dystonia. A multimodal physiotherapy program was applied in nine studies [30–32, 34–39], one study of high and four of moderate methodological quality. The total number of participants included in RCTs was 132. Multimodal physiotherapy programs combine multiple of the abovementioned modalities. Tassorelli et al. [32] conducted a RCT in 40 patients with CD. A 2-week physiotherapy program combined with BoNT/A and EMG biofeedback showed no beneficial effect on the severity of CD measured by the TWSTRS and Tsui scale, yet patients experienced significantly less pain, increased functioning and the effect of BoNT/A lasted significantly longer, requiring a lower dose for the following injection. In a case series of six patients [36], a 4-week physiotherapy program of relaxation, active exercises to strengthen the antagonist muscles, training of balance, coordination and body perception resulted in decreased pain and disability and an improved quality of life. The improved QoL remained after 6 months of follow-up. A 6-week program of stretching, active exercises and TENS in combination to BoNT/A led to decreased disability, a significant decrease of rotational deviation of the head and improved fine motor hand function [37]. In the RCT of Queiroz et al. [38], the control group was treated with BoNT/A and the intervention group was treated with BoNT/A combined with a physiotherapy program consisting of motor learning exercises, stretching, active and passive mobilization of the cervical spine and functional electrical stimulation of the antagonist muscles. After the 4-week program, both groups showed a significant decrease of the severity of CD on the TWSTRS, showing the positive results of the BoNT/A injection. Yet the patients who received the multimodal physiotherapy program showed also a significant decrease on the disability and pain subscale of the TWSTRS. Secondly, both physical and mental health on the SF-36 significantly improved.
In the recent RCT of Boyce et al. [39], the intervention group received whole body relaxation and active exercises to increase the muscle strength of the antagonist muscles to induce a normal head position. This was compared to the control group who received only whole body relaxation. Patients treated with BoNT/A as well as patients receiving no other treatment were included. After a period of 12 weeks with eight treatment sessions, no positive effect of active exercises was found; only a trend towards better scores on the TWSTRS and lower depression were observed. A multimodal physiotherapy program adjunct to BoNT/ A injections may improve head position, decrease pain levels in patients with CD and improve functioning in every day activities on short term.
Discussion The aim of this systematic literature study was to evaluate the effectiveness of physiotherapy alone or in addition to BoNT/A injections in the treatment of CD. We identified 16 studies reporting the effectiveness of physiotherapy in CD patients. Only six RCTs were included, one clinical trial with a cross-over design, three case series and six case reports. The methodological quality of the RCTs was moderate to high with a high risk of selection bias due to a lack of blinding of both patients and observers as well as randomization. And in most of the studies, no follow-up period was reported. EMG biofeedback was applied in five studies. There is limited, low-quality evidence for the application of EMG biofeedback training as a solitary treatment for CD. Three trials of moderate methodological quality evaluated the effect of physiotherapy as an adjuvant treatment to BoNT/A injections [32, 37, 38]. Based on their findings, we can conclude that a multimodal physiotherapy treatment adjunct to BoNT/A injections may improve head position, decrease pain levels in patients with CD and improve functioning in every day activities on short term. One trial of high methodological quality showed a trend to improved scores on the TWSTRS [39]. The lack of significance might be a result of similarities in both treatment groups. Whole body relaxation was applied in both treatment groups. In the active group, it was combined with an active exercise program. The control group received no additional treatment. Given the fact that mental and physical load may aggravate the symptoms of CD, it is possible that whole body relaxation in itself has a beneficial effect on the symptoms of CD. The content of multimodal physiotherapy treatment varied, including active exercises, muscular elongation techniques or stretching, massage, relaxation, active and
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passive mobilization of the cervical spine and electrotherapy such as EMG biofeedback, TENS, or electrical stimulation of the antagonist muscles. Most active exercises are based on the therapy program of Bleton [40], in which muscles with the opposite function of the direction in which dystonic muscles pull the head are strengthened. These types of active exercises are included in multimodal therapy programs as well as other postural exercises aiming to correct the impaired position of the head. Regardless of the type of exercises performed, there is some evidence that an active approach can ameliorate the symptoms of cervical dystonia. The number of treatments also varied widely and ranged from eight to 36. Consequently, it is currently not clear how many treatment sessions should be prescribed. In the description of the patient population, gender, age and illness duration were most frequently reported. Mean age varied from 32.5 to 57.8 years and the mean illness duration varied from 1.8 to 16 years, which may indicate that patients who are recently diagnosed as well as patients with a long history of CD might benefit from an additional multimodal physiotherapy treatment. However, it would also be interesting to know whether the severity of CD plays a role in the treatment effect or if the effect of a multimodal physiotherapy program is depended on the severity of CD. Only three studies reported the severity of CD measured by the TWSTRS with a mean score varying from 34.6 to 49 [36, 38, 39]. We suggest the use of a severity scale such as the TWSTRS or CDIP-58 when describing future patient samples. This is in line with the recommendations by Albanese et al. [41]. This review has strengths and limitations. We focused on the treatment of cervical dystonia solely and two reviewers independently assessed the methodological quality of the included articles in order to reduce the risk of bias. We were able to select recently published RCTs [37– 39], which provided more information of controlled intervention physiotherapy treatments consisting of active exercises, stretching and biofeedback or transcutaneous electrical stimulation especially in combination with BoTN/A injections. In this systematic literature search, we only searched two databases and only in English, German, Dutch or French. It is, therefore, possible that we have missed some clinical studies. We would like to stipulate that the findings should be interpreted with caution. The limited number of RCTs, the limited methodological quality and the total of 132 patients reported in the RCTs diminish the power of recommendations that can be made. The rather low prevalence of idiopathic cervical dystonia [3, 4] may be the reason for the low number of participants. The content of the multimodal
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physiotherapy interventions and active exercises differed widely among the studies, making it difficult to identify the most effective component. Further research should address randomization, doubleblinding and should consider working multicentrical to enlarge the study population. Also, more objective parameters concerning the patient population should be reported. For instance, the diagnostic criteria for CD should be reported along with objective parameters such as the Cervical Dystonia Impact Profile and the Toronto Western Spasmodic Torticollis Rating Scale. This can be used to describe included samples and therapy progress in a more uniform manner.
Conclusions Regardless of the central neurological etiology of cervical dystonia, the evidence that a multimodal physiotherapy program consisting of active exercises, stretching and relaxation in addition to botulinum toxin treatment induces beneficial effects on the pain and disability of patients with cervical dystonia on short term is growing. Additional high-quality trials are needed before firm recommendations can be made. Acknowledgments We would like to thank Birgit Daalderop for helping with the data collection. Funding
University of Antwerp (BDHG/3449/18.12.202).
Conflicts of interest On behalf of all authors, the corresponding author states that there is no conflict of interest.
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