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  • 8/10/2019 Does Evidence Support Physiotherapy Management of Adult Complex Regional Pain Syndrome Type One a Syste

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    Review

    Does evidence support physiotherapy management of adult

    Complex Regional Pain Syndrome Type One? A systematic review

    Anne E. Daly a,b,*, Andrea E. Bialocerkowski b

    a Department of Physiotherapy, Austin Hospital, P.O. Box 5555, Heidelberg 3084, Victoria, Australiab School of Physiotherapy, The University of Melbourne, Parkville 3010, Victoria, Australia

    a r t i c l e i n f o

    Article history:Received 26 November 2007

    Received in revised form 19 March 2008

    Accepted 1 May 2008

    Available online 10 July 2008

    Keywords:

    Complex Regional Pain Syndrome Type One

    Physiotherapy

    Systematic review

    Critical review form

    a b s t r a c t

    Objective: To source and critically evaluate the evidence on the effectiveness of Physiotherapy to manageadult CRPS-1.

    Design: Systematic literature review.

    Methods: Electronic databases, conference proceedings, clinical guidelines and text books were searched

    for quantitative studies on CRPS-1 in adults where Physiotherapy was a sole or significant component of

    the intervention. Data were extracted according to predefined criteria by two independent reviewers.

    Methodological quality was assessed using the Critical Review Form.

    Results: The search strategy identified 1320 potential articles. Of these, 14 articles, representing 11 stud-

    ies, met inclusion criteria. There were five randomised controlled trials, one comparative study and five

    case series. Methodological quality was dependent on study type, with randomised controlled trials being

    higher in quality. Physiotherapy treatments varied between studies and were often provided in combina-

    tion with medical management. This did not allow for the stand-alone value of Physiotherapy to be

    determined. Heterogeneity across the studies, with respect to participants, interventions evaluated and

    outcome measures used, prevented meta-analysis. Narrative synthesis of the results, based on effect size,

    found there was good to very good quality level II evidence that graded motor imagery is effective in

    reducing pain in adults with CRPS-1, irrespective of the outcome measure used. No evidence was foundto support treatments frequently recommended in clinical guidelines, such as stress loading.

    Conclusions: Graded motor imagery should be used to reduce pain in adult CRPS-1 patients. Further, the

    results of this review should be used to update CRPS-1 clinical guidelines.

    2008 European Federation of Chapters of the International Association for the Study of Pain. Published

    by Elsevier Ltd. All rights reserved.

    1. Introduction

    Complex Regional Pain Syndrome Type One (CRPS-1), which is

    characterized by pain, swelling, autonomic and motor disturbances

    in the absence of a peripheral nerve injury, is a disabling and dis-

    tressing condition that is difficult to treat effectively. Many treat-

    ments have been developed to manage this condition due to the

    spectrum of symptoms and an incomplete understanding of the

    pathophysiology behind CRPS-1. These treatments include allied

    health therapies, such as physiotherapy, complementary and alter-

    native medicine, medical, pharmacological and psychological ap-

    proaches (Wilson et al., 2005).

    There is some evidence to support the use of pharmacological

    approaches in the management of CRPS-1. For example, anticon-

    vulsants, sodium channel blockers, oral corticosteroids, calcium

    regulating drugs and free radical scavengers and the management

    of neuropathic pain strategies (such as the use of antidepressants,

    anticonvulsants, NMDA-receptor antagonists) appear to reduce

    CRPS-1 symptoms (Baron, 2005; Cepeda et al., 2002). However,

    other treatment approaches, such as spinal cord stimulation and

    modulation of the sympathetic nervous system via surgical sympa-

    thectomy, a-adrenergic blockade, intravenous regional block and

    local anaesthetic block have equivocal evidence (Baron, 2005;

    Cepeda et al., 2002; Nelson and Stacey, 2006; Stanton-Hicks,

    2006), based on the results of systematic reviews (Cepeda et al.,

    2002; Forouzanfar et al., 2002; Jadad et al., 1995) and meta-analy-

    ses (Kingery, 1997; Perez et al., 2001).

    Physiotherapy is advocated in CRPS clinical guidelines (Harden

    et al., 2006c; Stanton-Hicks et al., 2002), textbooks on pain

    (Giamberardino, 2002; Harden et al., 2001; Janig and Stanton-

    Hicks, 1996; Justins, 2005; McMahon and Koltzenburg, 2005;

    Wilson et al., 2005) and narrative CRPS reviews (Baron and Janig,

    2004; Berthelot, 2006; Birklein, 2005; Dommerholt, 2004; Harden

    et al., 2006a; Quisel et al., 2005; Sharma et al., 2006; Wasner et al.,

    2003). Despite this advocacy, the role of physiotherapy in the

    1090-3801/$36.00 2008 European Federation of Chapters of the International Association for the Study of Pain. Published by Elsevier Ltd. All rights reserved.doi:10.1016/j.ejpain.2008.05.003

    * Corresponding author. Address: Department of Physiotherapy, Austin Hospital,

    P.O. Box5555, Heidelberg 3084, Victoria, Australia. Tel.: +61 3 94965461; fax: +613

    9457 6326.

    E-mail address:[email protected](A.E. Daly).

    European Journal of Pain 13 (2009) 339353

    Contents lists available at ScienceDirect

    European Journal of Pain

    j o u r n a l h o m e p a g e : w w w . E u r o p e a n J o u r n a l P a i n . c o m

    mailto:[email protected]://www.sciencedirect.com/science/journal/10903801http://www.europeanjournalpain.com/http://www.europeanjournalpain.com/http://www.sciencedirect.com/science/journal/10903801mailto:[email protected]
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    treatment of CRPS has not been rigorously evaluated using system-

    atic methodologies. Therefore, physiotherapy for CRPS is not pres-

    ently underpinned by research evidence (Cepeda et al., 2002).

    Although the scope and complexity of physiotherapy practice

    varies world wide, physiotherapy can be thought of as

    The treatment of disorders with physical agents and methods

    (Anderson, 2002)

    Physical agents and methods include manual therapy, electro-

    therapy, transcutaneous electrical nerve stimulation, massage

    and therapeutic exercise. Often these treatments are contextua-

    lised by theories, such as operant behavioural theory (Fordyce,

    1976; Main et al., 2002), cognitive behavioural therapy (Main

    et al., 2002; Turk et al., 1983), fear avoidance and graded exposure

    in vivo theory (Vlaeyen et al., 2001, 2002). Additionally, empower-

    ment and education of the patient underpin the delivery of physio-

    therapy treatment (Australian Physiotherapy Association, 2004).

    Althoughsome physiotherapy evidence has been includedin the

    work by Forouzanfar et al. (2002), the entire body of physiotherapy

    evidence has not been synthesized. Therefore, the objective of this

    systematic review was to source, critically evaluate and synthesize

    current physiotherapy research evidence for the treatment of adult

    CRPS-1. The results of this study could then be used to make evi-

    dence-based recommendations regarding the physiotherapy treat-

    ments for CRPS-1 and to update CRPS treatment guidelines.

    2. Method

    2.1. Search strategy

    An electronic search was performed in September, 2007 cover-

    ing the previous two decades of the following databases: CINAHL

    (19872007), Medline (19872007), Embase (19872007), ISI

    Web of Science (19872007), and the following evidence-based

    practice resources; Cochrane Library (19872007) (The Cochrane

    Collaboration, 2007), Turning Research into Practice (TRIP) Data-base (complete site) (TRIP Database, 2007), Physiotherapy Evi-

    dence Database (PEDro) (complete site) (Centre for Evidence

    Based Physiotherapy, 2007), Joanna Briggs Institute (complete site)

    (Joanna Briggs Institute, 2007). The websites for these databases

    are included in the reference list.

    Key words used to describe CRPS-1 and physiotherapy were

    listed. Multiple keywords describing CRPS-1 were used as different

    terms were used prior to 1994 when the International Association

    for the Study of Pain (IASP) introduced the term Complex Regional

    Pain Syndrome Type One (Merskey and Bogduk, 1994). Key words

    describing physiotherapy were also numerous reflecting both the

    range of physiotherapy interventions and the terminology used to

    describe such interventions (Table 1). Where possible, key words

    were used to identify relevant Medical Subject Headings (MeSH)

    thatwere thenexploded. To gaina listof potentially relevantpapers,

    the CRPS-1 keywords were combined using OR. This strategy was

    repeated for the physiotherapy keywords. To identify papers on

    physiotherapy interventionsfor CRPS-1the twogroupsof keywords

    were combined using AND. Truncation and wild card symbols

    were usedto retrieve all relevantkeywords. In non-MeSH databases

    the following CRPS-1 keywords were used: Complex Regional Pain

    Syndrome, CRPS, Reflex SympatheticDystrophy, RSDand were com-

    bined using OR. The term Complex Regional Pain Syndrome was

    used in databases that would only accept one term.

    In addition, secondary search strategies were performed by con-

    ducting hand searches of

    Reference lists of textbooks on pain (Giamberardino, 2002; Jus-

    tins, 2005; McMahon and Koltzenburg, 2005) and CRPS (Harden

    et al., 2001; Janig and Stanton-Hicks, 1996; Wilson et al., 2005).

    Proceedings of the 11th World Congress on Pain (Flor et al.,

    2006).

    Reference lists of all papers meeting the inclusion criteria.

    Internet searches of 30 contributors to three CRPS clinical

    guidelines were undertaken (Harden et al., 2006b; Harden et al.,

    2006c; Stanton-Hicks et al., 2002) to identify other relevant

    publications.

    2.2. Inclusion criteria

    All titles and abstracts were read to identify relevant papers. Pa-

    pers were included in this systematic review if they met the inclu-

    sion criteria listed in Table 2.The type of outcome measure used to

    evaluate the effect of physiotherapy was not an inclusion criterion

    for this review. Currently, consensus is lacking with regards to a

    gold standard of outcome measure for both physiotherapy andCRPS-1. Furthermore, as physiotherapy generally aims to affect

    multiple outcomes such as pain intensity, strength, weight bearing,

    swelling, activity limitation and participation restriction this al-

    lowed for a broad consideration of the effectiveness of

    physiotherapy.

    When there was uncertainty regarding the eligibility of the pa-

    per from the abstract, the full text version of the paper was re-

    trieved and evaluated against the inclusion criteria. The full text

    version of all papers that met the inclusion criteria were retrieved

    for quality assessment and data extraction.

    Table 1

    Keywords used to develop the search strategy

    Population Intervention

    Complex regional pain syndrome Physiotherapy Therapeutic exercise

    CRPS Physical therapy Exercise

    Complex regional pain syndrome type one Manual therapy Mirror visual feedback

    CRPS-1 Manipulative Physiotherapy Mirror therapy

    Reflex Sympathetic Dystrophy Manipulative therapy Graded motor imagery

    RSD Hand therapy Graded imagery

    Reflex Sympathetic Dystrophy syndrome Rehabilitation Stress loading program

    RSDS Electrotherapy Stress loading

    Sympathetic Dystrophy Heat therapy Scrub and carry

    Neuropathic pain Cryotherapy Massage

    Shoulder hand syndrome Ultrasound Sensory motor integration

    Sudecks atrophy Transcutaneous electrical nerve stimulation Water exercise

    Algodystrophy TENS Aquatic Physiotherapy

    Low frequency electrical cutaneous therapy Aquatic therapy

    Magnetic Aquatic exercise

    Feldenkrais Hydrotherapy

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    2.3. Quality assessment

    Critical appraisal of each included study was conducted by

    determining:

    The level of evidence on the Australian National Health and

    Medical Research Council (NHMRC) Hierarchy of Evidence (Aus-tralian National Health and Medical Research Council, 1999)

    (Table 3). This provides a broad indication of bias based on study

    design. Studies higher on the hierarchy potentially contain less

    bias than those that are lower on the hierarchy.

    The methodological quality of the study using the Critical

    Review Form Quantitative studies (Law et al., 1998a). This tool

    can be used to appraise all types of quantitative studies ranging

    from RCTs to case series. Thus all quantitative studies on phys-

    iotherapy management of CRPS-1 were included in this review

    and evaluated for quality using the same tool. This made the

    quality results comparable between the different study designs

    (Petticrew, 2003). Standardised guidelines on the interpretation

    and scoring of each item were used (Law et al., 1998b). Items

    were scored as 1 (completely fulfils the criterion) or 0 (doesnot completely fulfil the criterion). The scores of the 16 closed

    ended questions were tallied to provide an overall score of qual-

    ity where the maximum score of 16 indicated excellent quality

    (Bialocerkowski et al., 2005). Two researchers independently

    scored the studies and where disagreement occurred, consensus

    was achieved by discussion. Quality scores were arbitrarily

    divided into five categories: poor (score6 8), fair (score = 9

    10), good (score = 1112), very good (score = 1314) and excel-

    lent (score = 1516).

    The Critical Review Form Quantitative studies (Law et al.,

    1998a) includes 17 of the 22 items that are contained in the

    CONSORT statement (Altman et al., 2001; Moher et al., 2001).

    It does not include items one (study design stated in title or

    abstract), eight, nine and 10 (randomisation: sequence genera-

    tion, allocation concealment and implementation respectively)

    or 19 (adverse events). The CONSORT statement was not

    designed to evaluate methodological quality (Altman et al.,

    2001). However, in this review, it was noted whether these five

    CONSORT criteria were fulfilled by the RCTs. This would providethe reader with further methodological quality information.

    2.4. Data extraction

    Data were extracted by the authors using the PICO approach

    (Stone, 2002)

    Participants: area of involvement of CRPS, age, sex, duration of

    symptoms, type of referral source and diagnostic criteria.

    Interventions: type, intensity, duration, in combination or stand-

    alone physiotherapy.

    Comparison: to another treatment, no treatment or usual

    treatment. Outcomes: domains and tools used to measure the effects of the

    intervention. These were grouped according to the World Health

    Organisation (WHO) International Classification of Functioning,

    Disability and Health: abnormalities of body function and struc-

    ture; activity limitation; and participation restriction (World

    Health Organisation, 2001). Additional domains included quality

    of life and cost effectiveness.

    Data on the effectiveness of the physiotherapy treatment were

    also extracted for each study. To determine the effect of the

    physiotherapy treatment on each outcome measure, the mean

    and 95% confidence interval for the between-group differences

    Table 3

    Study design, based on the Australian National Health and Medical Research Council Hierarchy of Evidence (Australian National Health and Medical Research Council, 1999)

    Level Definition Studies

    I Evidence obtained from a systematic review of all relevant randomised controlled trials

    II Evidence obtained from at least one properly-designed randomised controlled trial Durmus et al. (2004), Moseley (2004, 2005, 2006); Oerlemans

    studya

    III-1 Evidence obtained from well-designed pseudo-randomised controlled trials (alternate

    allocation or some other method)

    III-2 Evidence obtained from comparative studies (including systematic reviews of such studies)

    with concurrent controls and allocation not randomised, cohort studies, casecontrol studies,

    or interrupted time series with a control group

    McCabe et al. (2003)

    III-3 Evidence obtained from comparative studies with historical control, two or more single arm

    studies, or interrupted time series without a parallel control group

    IV Evidence obtained from case series, either post-test or pre-test/post-test de Jong et al. (2005), Pleger et al. (2005), Robaina et al. (1989),

    Singh et al. (2004), Watson and Carlson (1987)

    a Oerlemans et al. (1999a,b, 2000b), Severens et al. (1999).

    Table 2

    Inclusion criteria used in the systematic review

    Criterion Justification

    1 English language Major journals in this area are published in this language

    2 19872007 Twenty years captures the most frequently used physical modalities in clinical practice today

    3 Studied the outcome of Physiotherapy alone or in combination

    with other medical or psychological therapies

    As interdisciplinary management of CRPS-1 is considered optimal (Stanton-Hicks et al., 2002),

    studies that involved Physiotherapy in combination with other interventions were included

    4 Humans over 18 years of age This increased the homogeneity of participants between the studies

    5 Stated diagnostic criteria based on clinical presentation Diagnostic criteria must be stated so that the reader can determine how likely it was that astudy population actually had CRPS 1 and how similar the study population was compared to

    their own CRPS 1 population

    6 Quantitative study design including randomised controlled trials

    (RCTs), non-randomized clinical trials or case series

    Study designs other than RCTs were included in this review as they provide complementary

    and relevant clinical detail to the current state of our knowledge and its limitations (Barton,

    2000; Norris and Atkins, 2005). Case studies were not included because of the low level of

    evidence they provide

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    was calculated for RCTs and comparative studies, based on the

    results provided in each article (Herbert, 2000). Moreover the

    mean change between pre and post treatment (and 95% confi-

    dence intervals) were calculated for the case series. Pain reduc-

    tion of more than 20%, irrespective of the measurement tool

    used, was considered clinically worth-while (Farrar et al., 2001;

    Ferreira et al., 2002). It was expected that there would be heter-

    ogeneity in participants, interventions, comparisons and out-

    comes. Therefore the results of the studies were synthesized in

    a narrative format.

    3. Results

    3.1. Search strategy yield

    Initially 1320 hits were gained from database and secondary

    search strategies. After review of the titles and abstracts, duplicates

    and those articles that did not meet the inclusion criteria were re-

    moved. After reviewing the full text of 180 articles, 166 articles

    were excluded as they did not meet the inclusion criteria. There-

    fore 14 articles were included in this systematic review (Fig. 1)

    (de Jong et al., 2005; Durmus et al., 2004; McCabe et al., 2003;

    Moseley, 2004, 2005, 2006; Oerlemans et al., 1999a,b, 2000b; Pleg-

    er et al., 2005; Robaina et al., 1989; Severens et al., 1999; Singh

    et al., 2004; Watson and Carlson, 1987). However, four of these

    articles (Oerlemans et al., 1999a,b, 2000b; Severens et al., 1999)

    were derived from one large RCT but were published separately

    due to the diversity of outcomes measured. These articles will be

    referred to as the Oerlemans study. Consequently, 11 studies were

    included in this systematic review.

    3.2. Critical appraisal

    3.2.1. Hierarchy of evidence

    There were five RCTs (Durmus et al., 2004; Moseley, 2004, 2005,

    2006, Oerlemans study), one comparative study (McCabe et al.,

    2003), and five case series (de Jong et al., 2005; Pleger et al.,

    2005; Robaina et al., 1989; Singh et al., 2004; Watson and Carlson,

    1987)(Table 3).

    3.2.2. Methodological quality

    There was 98% agreement in scoring between the researchers

    conducting the systematic review. Disagreements were resolved

    by discussion. Variation in methodological quality was noted (Ta-

    ble 4), with scores ranging from two (Robaina et al., 1989) to 13

    (Moseley, 2006; Oerlemans et al., 2000b) (mean = 10/16). The

    majority of papers were good in quality (de Jong et al., 2005; Dur-

    mus et al., 2004; Moseley, 2004, 2005; Oerlemans et al., 1999a,b;

    Severens et al., 1999; Singh et al., 2004), two were poor (Robaina

    et al., 1989; Watson and Carlson, 1987), two were fair (McCabe

    et al., 2003; Pleger et al., 2005) and two were very good (Moseley,

    2006; Oerlemans et al., 2000b). No papers were rated as excellent.

    In general, the level II studies had higher quality scores than the le-

    vel III study, which was higher in quality than the level IV studies

    (mean = 12, 10, 8 points respectively).

    Table 4 provides details regarding the criteria that were fulfilled

    on the Critical Appraisal Form Quantitative studies (Law et al.,

    1998a). It demonstrates that only half the studies provided ade-

    quate detail to allow for reproduction of their interventions (crite-

    rion 10). Only two studies reported the reliability of all their

    measurement tools (criterion 9), one justified sample size (crite-

    rion 6), and no studies were free from major biases (criterion 4)

    or reported the validity of all their measurement tools (criterion 8).

    3.2.3. CONSORT criteria 1, 8, 9, 10 and 19

    Table 4 also provides details regarding the fulfillment of the

    CONSORT criteria. None of the RCTs complied with item nine by

    reporting the method used to implement the random allocation se-quence. OnlyMoseley (2004, 2005, 2006) complied with item 10

    by reporting who generated the allocation sequence, enrolled par-

    ticipants and assigned participants to their groups. No studies

    complied with item 19 by reporting whether there were any ad-

    verse events in any intervention group.

    3.2.4. Types of intervention

    In all studies physiotherapy was a major (Durmus et al., 2004;

    Moseley, 2004, 2005, 2006, Oerlemans study;Pleger et al., 2005;

    Singh et al., 2004) or sole (de Jong et al., 2005; McCabe et al.,

    2003; Robaina et al., 1989; Watson and Carlson, 1987) source of

    treatment. Details of the specific physiotherapy treatments evalu-

    ated are found inTable 5. In summary, physiotherapy consisted of

    Education (de Jong et al., 2005).

    Graded exposure in vivo to activities, movements and light

    touch (de Jong et al., 2005).

    Sensorimotor treatment: pain adapted exercises and desensitisa-

    tion activities (Pleger et al., 2005).

    Exercise: stretching, active range of motion (Durmus et al., 2004),

    water therapy (Singh et al., 2004), stress loading (Watson and

    Carlson, 1987).

    Mirror visual feedback: a mirror is placed between the affected

    and unaffected limb, obscuring the affected limb and exercises

    are performed while the participant looks at the image of the

    unaffected limb (McCabe et al., 2003).

    Graded motor imagery: a motor imagery program (MIP) where

    the subject recognises the laterality of a photograph of a handor foot, followed by imagining moving their painful limb into

    Review of title & abstract

    Removal of duplicates

    Ineligible if:

    Not published in English language

    Did not study Physiotherapy

    Review article, letter or comment

    Conference abstract or dissertation

    Articles retrieved

    n = 1320

    Not eligible n = 1140Potentially eligible

    n = 180

    Review of full text

    Ineligible if:

    Did not describe diagnostic procedures

    Physiotherapy was not a significant intervention

    Subjects aged < 18 years

    Eligible articles

    n = 14

    Individual studies

    n = 11

    Not eligible n = 166

    Fig. 1. Retrieval and review process.

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    Table 4

    Study quality using the Critical Review Form Quantitative studies (Law et al., 1998a,b) and the CONSORT statement ( Moher et al., 2001)

    Criterion Critical

    Review Form

    de Jong et al.

    (2005)

    Durmus et al.

    (2004)

    McCabe et al.

    (2003)

    Moseley

    (2004)

    Moseley

    (2005)

    Moseley

    (2006)

    Oerlemans

    studyaPleger et al.

    (2005)

    Robaina et a

    (1989)

    1 Purpose clearly stated 1 1 1 1 1 1 1 1 0

    2 Literature review

    relevant

    1 1 1 1 1 1 1 1 0

    3 Study design

    appropriate to study

    aims

    1 1 1 1 1 1 1 1 0

    4 No biases present 0 0 0 0 0 0 0 0 0

    5 Sample described in

    detail

    1 1 1 1 1 1 1 0 0

    6 Sample size justified 0 0 0 0 0 1 0 0 0

    7 Informed consent

    gained

    1 1 0 1 1 1 1 1 0

    8 Validity of outcome

    measures used

    0 0 0 0 0 0 0 0 0

    9 Reliability of outcome

    measures used

    0 0 1 0 0 0 0 0 0

    10 Intervention described

    in detail

    1 1 1 1 1 1 0 0 0

    11 Statistical reporting of

    results

    1 1 0 1 1 1 1 1 0

    12 Appropriate statistical

    analysis

    1 1 1 1 1 1 1 1 0

    13 Clinical importance

    reported

    1 1 1 1 1 1 1 1 1

    14 Appropriate

    conclusions

    1 1 1 1 1 1 1 1 0

    15 Clinical implications

    reported

    1 1 1 1 1 1 1 1 1

    16 Study limitations

    acknowledged

    1 1 0 1 1 1 1 0 0

    Total 12 12 10 12 12 13 11 9 2

    Quality categoryb Good Good Fair Good Good Very good Good Fair Poor

    Criterion CONSORT statement

    1 Study design stated in

    title or abstract

    U U U U U

    8 Randomisation:

    sequence generation

    U U U U U

    9 Randomisation:

    allocation concealment

    10 Randomisation:

    implementation

    U U U

    19 Adverse events

    a Oerlemans et al. (1999a,b), Severens et al. (1999); quality score = 11,Oerlemans et al. (2000b),criteria 6 and 9 also fulfilled, quality score = 13, U = criterion fulfilled, = cb Quality category: poor (score 6 8), fair (score = 910), good (score = 1112), very good (score = 1314) and excellent (score = 1516).

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    Table 5 (continued)

    Author Participants Intervention Outcomes

    n Sample characteristics Diagnostic criteria Treatment Control Outcome instr

    Oerlemans

    study1,2,3,4135 RSD UL< 1 year duration recruitment:

    outpatient clinics, University Hospitals

    in Nijmegan or Amsterdam, The

    Netherlands, female = 95, mean age 53,

    right UL 45%, mean duration 3.6

    Months

    Veldman et al.

    (1993)

    Physiotherapy or Occupational

    Therapy with specific treatment

    objectivese and fixed protocol medical

    treatment

    Social workf in

    addition to fixed

    protocol medical

    treatment

    Impairmen

    ation of Per

    Active rang

    of all UL joi

    Sensory los

    Grip streng

    Pain inten

    pain at pre

    effort, MPQ

    Impairmen

    score (ISS)3

    Disability: M

    RASQ3, Radb

    Handicap:

    (SIP)3,4

    Cost effect

    tion, differe

    tal costs,

    visits, trave

    and work

    tiveness4

    1No signific

    OT and cont

    to permane

    follow-up.

    and to a less

    value over Sp< 0.05, O

    p< 0.10) a

    increased j

    (p< 0.05) b

    months. Qu

    the PT grou3Adjuvant P

    extent OT (

    relevant th

    ment which

    low-up. 4P

    effective t

    CRPS-1 com

    (SW). Costs

    treatment a

    other medic

    Pleger et al.

    (2005)

    6 CRPS-1of the whole hand, including all

    digits, recruitment: BG-Kliniken,

    Bergmannsheil, Bochum, Germany left

    UL = 4, duration range 152 months

    Bruehlet al. (1999) Pain adapted sensorimotor treatment

    protocol (graded desensitisation and

    motor tasks) and usual drug therapy

    No Pain intens

    now and ov

    Immobility

    points

    Cortical m

    (fMRI)

    Peripheral

    crimination

    Graded sen

    and 6 mont

    intensity (p

    tactile discr

    regaining o

    (p = 0.02) a

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    the position in the photograph. This is progressed to moving

    both limbs into the position while observing the unaffected limb

    in a mirror which obscures the affected limb (Moseley, 2004,

    2005, 2006). Physical therapy: no description provided (Singh et al., 2004).

    Pain management techniques including improving pain control,

    optimizing coping, relaxation, connective tissue massage,

    improving skills, extinguishing pain source, developing compen-

    satory skills (Oerlemans study).

    Transcutaneous electrical nerve stimulation (Robaina et al.,

    1989).

    Electromagnetic field treatment (Durmus et al., 2004).

    3.2.5. Use of control groups

    Control interventions varied between the Level II studies, and

    included the

    Maintenance of existing medical and physiotherapy manage-

    ment (Moseley, 2004).

    Implementation of standard medical and physiotherapy man-

    agement including a home exercise program at the same inten-

    sity as the experimental group (Moseley, 2006).

    Use of the same drug and exercise program as the experimental

    group and a sham form of electromagnetic field treatment (Dur-

    mus et al., 2004).

    Use of the same medical management as the experimental

    group and added passive attention via social work sessions (Oer-

    lemans study).

    3.2.6. Outcome measures

    There was great variability in outcome measurement across the

    studies, in terms of the number and type of outcome measures

    used and the number of occasions on which they were used

    (Table 6). The majority of studies used outcome instruments that

    addressed disorders of body structure or function (World Health

    Organisation, 2001). Only five studies measured other constructssuch as activity limitation (de Jong et al., 2005; Moseley, 2005,

    2006; Oerlemans et al., 2000b; Severens et al., 1999; Singh et al.,

    2004), participation restriction (Singh et al., 2004), cost effective-

    ness (Oerlemans et al., 2000b; Severens et al., 1999), quality of life

    (Oerlemans et al., 2000b; Severens et al., 1999) or treatment

    credibility and expectancy (de Jong et al., 2005). Pain was the most

    frequently measured variable; however it was not evaluated in a

    consistent manner across the studies. For example, the verbal rat-

    ing scale, Neuropathic Pain Scale (NPS), the intensity score of the

    NPS, the McGill Pain Questionnaire, the 11-point numeric rating

    scale and various visual analogue scales were used to quantify a

    change in pain following physiotherapy treatment. Moreover,

    measurement periods were variable, ranging from two weeks

    (Moseley, 2004) to three years (Robaina et al., 1989), but often rep-resented the immediate and longer term effects of physiotherapy.

    3.3. Participants

    Participant characteristics varied across the studies. Three stud-

    ies included participants with both upper and lower limb CRPS-1

    (de Jong et al., 2005; McCabe et al., 2003; Moseley, 2006), whereas

    the remaining eight studies included participants with only upper

    limb CRPS-1. Mean age ranges varied from 33 years (McCabe et al.,

    2003) to 53 years (Oerlemans study), although three studies did

    not provide this detail (de Jong et al., 2005; Moseley, 2006; Pleger

    et al., 2005). Males and females were recruited in varying

    proportions ranging from 100% female (de Jong et al., 2005) to

    37% female (Watson and Carlson, 1987) (mean = 67%). Average

    duration of symptoms varied from 3.6 months (Oerlemans study)

    Table 6

    Classification of outcomes according to the World Health Organisations 2001 International Classification of Functioning, Disability and Health (WHO, 2001)

    Concept ICF 2001 terminology Domain identified in

    CRPS-1 studies

    Tools used in CRPS-1 studies

    Disorder of body structure

    or function

    Abnormality of body

    structure and function

    Pain intensity and

    quality

    Visual analogue scale, 11 point rating scale, Verbal pain scale, Neuropathic pain scale,

    McGill pain questionnaire, Diagnostic criteria

    Weight bearing

    tolerance

    Weight scale and timer

    Pressure tolerance Force gauge and timerSensation 2 point discriminator

    Strength Jamar dynamometer

    Range of motion Goniometer

    Swelling Water displacement volumeter, Digital circumference tape measure

    Dexterity Radboud dexterity test, JebsenTaylor test

    Limb laterality

    recognition

    Response time

    Autonomic changes Diagnostic criteria, Resting sweat output conductivitycell, Muscle blood flow blood

    pressure cuff, Point temperature infrared thermography

    Psychological and

    behavioural attributes

    CES-D scale, Pain locus of control, Pain cognitions questionnaire, Pain situations

    questionnaire, Tampa scale for Kinesiophobia, Photograph series of daily activities

    Other Three phase bone scan, Biochemical markers, Functional MRI, Impairment level

    sumscore, AMA Evaluation of Permanent Impairment

    Abnormality in function at

    the level of the person

    Activity limitation Radboud skills questionnaire, Walking stairs questionnaire, Rising and sitting down

    questionnaire, Self rating on 11 point scale of present ability to complete tasks,

    Modified Greentest, Baltimore therapeutic work simulator test

    Inability to fulfill normalroles Participation restriction Goal achievement Diary, Visual analogue scale, Baltimore therapeutic work simulator

    Cost effectiveness Differential cost

    analysis

    Hospital costs, patient diary of medical visits, travel, medications, home care and work

    absence

    Treatment

    effectiveness

    Impairment level sumscore, Modified Greentest, Sickness impact profile

    Quality of life Sickness impact profile

    Treatment credibility and

    expectancy

    Visual analogue scale

    MRI, magnetic resonance imaging, AMA, American Medical Association, CES-D, Centre for Epidemiological Studies Depression.

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    to 36 months (de Jong et al., 2005) (mean = 11.4 months, n= 10

    studies).

    Sample sizes in the studies ranged from six (Pleger et al., 2005)

    to 135 (Oerlemans study) (mean = 56). Subjects were recruited

    from University hospitals and specialist clinics in all studies with

    Moseley also recruiting participants from Physiotherapy depart-

    ments and local General Practice clinics (Moseley, 2004, 2005,

    2006). All studies listed or referenced their diagnostic criteria forCRPS-1 (Table 7). Most frequently the IASP criteria (Merskey and

    Bogduk, 1994) or the proposed modified research diagnostic crite-

    ria for CRPS-1 (Bruehl et al., 1999), were used(Durmus et al., 2004;

    McCabe et al., 2003; Moseley, 2004, 2005, 2006; Pleger et al., 2005;

    Singh et al., 2004).

    3.4. Effectiveness of physiotherapy

    Data gained from the RCTs could not be pooled because of the

    heterogeneity of the outcome measures and comparison groups.

    Tables 8a and 8billustrate the effect sizes of the studies included

    in this systematic review. Effect sizes could not be calculated for

    two case studies (Robaina et al., 1989; Singh et al., 2004) and

    95% confidence intervals could not be calculated for a furthertwo studies (Watson and Carlson, 1987; Moseley, 2005).

    Table 8a illustrates that graded motor imagery is more effec-

    tive than usual physiotherapy plus medical management in

    reducing pain, irrespective of the pain outcome measure used

    (Moseley, 2004, 2006). A statistically significant reduction in pain

    intensity occurred immediately after a six week program of

    graded motor imagery (Moseley, 2004, 2006) and this effect was

    maintained up to six months following treatment (Moseley,

    2004, 2006). These results are clinically relevant, as pain was re-

    duced on average by more than 20 points on a 100 point scale

    (Farrar et al., 2001; Ferreira et al., 2002). Graded motor imagery

    also reduced swelling in the fingers (Moseley, 2004), however it

    is difficult to interpret the magnitude of effect size as a clinically

    important change for this variable has not been determined. Theorder in which the components of graded motor imagery are pre-

    sented affect the magnitude of pain reduction, with laterality rec-

    ognition followed by imagined movements and mirror

    movements producing the greatest pain reduction (Table 8b)

    (Moseley, 2005). The Oerlemans study found statistically signifi-

    cant changes in the impairment sum score, temperature differ-

    ence between the hands, and pain (measured by the McGill Pain

    Questionnaire), such that physiotherapy and medical manage-

    ment was more effective than both occupational therapy andmedical management and social work and medical management.

    However, it is likely that these changes were not clinically signif-

    icant due to their small effect sizes.

    The data contained in Table 8b illustrate that significant

    changes between pre and post measurements were gained for

    two types of physiotherapy. Sensorimotor treatment reduced pain

    and increased tactile discrimination (Pleger et al., 2005) whereas a

    graded exposure in vivo program significantly reduced pain related

    fear and pain disability (de Jong et al., 2005). Mirror visual feed-

    back appears to reduce the perception of pain in early CRPS-1

    and stiffness in intermediate CRPS-1 (Table 8b) (McCabe et al.,

    2003) and an interdisciplinary pain management program in-

    creases function in CRPS-1 (Table 5) (Singh et al., 2004). There

    was no evidence to support the effectiveness of transcutaneouselectrical nerve stimulation (Robaina et al., 1989) and stress load-

    ing exercise (Watson and Carlson, 1987). The study results, as de-

    scribed by the authors and the statistical precision of the effect (p

    value) are listed inTable 5.

    4. Discussion

    4.1. Interpretation of the results

    This is the first systematic review that has considered the qual-

    ity of individual studies when synthesizing the results. The major

    findings were

    1. There is good to very good quality level II evidence that physio-therapy consisting of graded motor imagery, combined with

    Table 7

    Diagnostic criteria

    Diagnostic set Criterion 1 Criterion 2 Criterion 3 Sensitivity Specificity

    Watson and Carlson

    (1987)

    Pain out of proportion to

    trauma

    Swelling and vasomotor changes Stiffness

    Robaina et al. (1989) Pain Sudomotor and vasomotor alterations

    Veldman et al. (1993) 4 out of 5: Pain, Oedema

    Altered skin colour Altered

    skin temperature Reducedrange of motion

    Signs and symptoms must be present in

    area much larger than and distal to

    primary injury

    Signs and symptoms must be

    aggravated by activity of the limb

    0.65 0.85

    Perez et al. (2005)

    International

    Association for the

    Study of Pain criteria

    isMerskey and

    Bogduk (1994)

    Continuing pain, allodynia

    or hyperalgesia with which

    the pain is disproportionate

    to the inciting event

    Evidence at sometime of oedema,

    changes in skin blood flow or abnormal

    sudomotor activity in the region of the

    pain

    This diagnosis is excluded by the

    existence of conditions that would

    otherwise account for the degree of

    pain and dysfunction

    0.98 0.36

    Bruehl and Harden

    (2001)

    Proposed modified

    research diagnostic

    criteria for CRPS-1,

    Bruehl et al. (1999)

    Continuing pain that is

    disproportionate to any

    inciting event

    Must report at least 1 symptom in each

    of the 4 following categories:

    Must display at least 1 sign in 2 or more

    of the following categories:

    0.70 0.96

    Baron and Janig (2004)

    Sensory: reports of hyperaesthesia Sensory: evidence of hyperalgesia (to

    pinprick) and/or allodynia (to light

    touch)

    Vasomotor: reports of temperature

    asymmetry and/or skin colour changes

    and/or skin colour asymmetry

    Vasomotor: evidence of temperature

    asymmetry and/or skin colour changes

    and/or skin colour asymmetry

    Sudomotor/oedema: reports of oedema

    and/or sweating changes and/or

    sweating asymmetry

    Sudomotor/oedema : evidence of oedema

    and/or sweating changes and/or

    sweating asymmetryMotor/trophic: Reports of decreased

    range of motion and/or motor

    dysfunction (weakness, tremor,

    dystonia) and/or trophic changes (hair,

    nails, skin)

    Motor/trophic: Evidence of decreased

    range of motion and/or motor

    dysfunction (weakness, tremor,

    dystonia) and/or trophic changes (hair,

    nails, skin)

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    medical management is more effective than physiotherapy and

    medical management for upper or lower limb CRPS-1 (Moseley,

    2004, 2005, 2006). Graded motor imagery reduces pain inten-

    sity by a clinically relevant amount, and this reduction is main-

    tained for up to six months.

    2. There is good quality level II evidence that pain management

    physiotherapy combined with medical management is more

    effective than occupational therapy or social work combined

    with medical management in patients with upper limb CRPS-

    1 (Oerlemans study). However, the clinical relevance of this

    finding is questionable.

    3. There is good quality level II evidence that electromagnetic field

    treatment offers no added benefit to calcitonin and exercise in

    upper limb CRPS-1 patients (Durmus et al., 2004).

    In addition, there is good quality emerging evidence which sug-

    gests that a graded exposure in vivo program (de Jong et al., 2005)

    and a four week interdisciplinary outpatient treatment program

    (Singh et al., 2004) may be effective for CRPS-1. There is fair quality

    emerging evidence that a sensorimotor treatment program (Pleger

    et al., 2005) and mirror visual feedback (McCabe et al., 2003) may

    also be effective.

    Table 8a

    Effect of Physiotherapy treatments on outcomes measured on a continuous scale, produced from randomized controlled trials with intention to treat analyses

    Physiotherapy vs comparison (outcome measure and possible range)

    (reference time of follow-up)

    Sample size Quality score Mean change difference (95% CI)

    EMFT + C + Ex vs C+ Ex (pain intensity: 0100 mm VAS)

    Durmus et al. (2004) end 6 weeks Rx (pain at rest) 40 12 12 [0 to 23]

    Durmus et al. (2004) end 6 weeks Rx (pain with activity) 40 12 13 [1 to 26]

    EMFT+ C + Ex vs C + Ex (swelling: mls)

    Durmus et al. (2004) end 6 weeks Rx 40 12 6.5 [43.3 to 30.4]

    GMI vs PT + MM (pain intensity: 0100 VAS)

    Moseley (2006) end 6 weeks of Rx (av. pain over last 2 days) 51 13 12.9 [1.7 to 21.4]

    Moseley (2006) 6 month follow-up (av. pain over last 2 days) 51 13 20.5 [8.2 to 32.8]*

    GMI vs PT + MM (pain: NPS: 0100)

    Moseley (2004) end 6 weeks of Rx 13 12 22 [10.1 to 29.9]*

    Moseley (2004) 12 week follow-up 13 12 22 [13.4 to 30.6]*

    GMI vs PT + MM (pain intensity: NPS item on intensity: 010)

    Moseley (2004) end 6 weeks of Rx 13 12 3.0 [2.6 to 5.4]*

    Moseley (2004) 12 week follow-up 13 12 3.0 [2.8 to 5.6]*

    GMI vs PT + MM (finger circumference: mm)

    Moseley (2004) end 6 weeks of Rx 13 12 9.0 [2.3 to 15.7]*

    Moseley (2004) 12 week follow-up 13 12 10.0 [2.6 to 17.3]*

    GMI vs PT + MM (function: NRS: 010)

    Moseley (2006) end 6 weeks of Rx 51 13 1.6 [0.6 to 2.6]*

    Moseley (2006) 6 month follow-up 51 13 2.2 [1.1 to 3.3]*

    Rec + Im+ Mir vs Im + Rec + Im (pain: NPS: 0100)

    Moseley (2005) end of 6 weeks of Rx 20 12 5 [7.0 to 11]a

    Moseley (2005) 12 week follow-up 20 12 7 [3.0 to 17]a,*

    Rec+ Im + Mir vs Rec + Mir+ Rec (pain intensity: NPS: 0100)

    Moseley (2005) end of 6 weeks of Rx 20 12 10 [3 to 20]a,*

    Moseley (2005) 12 week follow-up 20 12 18 [5 to 25]a,*

    PT + MM vs OT + MM (impairment sum score: 050)

    Oerlemans studyb 12 month follow-up 135 13 1.6 [1 to 2.2]*

    PT + MM vs SW + MM (impairment sum score: 050)

    Oerlemans studyb 12 month follow-up 135 13 5.1 [4.6 to 5.6]*

    PT + MM vs OT + MM (temperature difference:C)

    Oerlemans studyb 12 month follow-up 135 13 0.1 [0.1 to 0.1]*

    PT + MM vs SW + MM (temperature difference: C)

    Oerlemans studyb 12 month follow-up 135 13 0.6 [0.6 to 0.6]*

    PT + MM vs OT + MM (pain intensity: 0100 VAS)

    Oerlemans studyb 12 month follow-up (pain with effort) 135 13 5.2 [3.3 to 7.1]

    PT + MM vs SW + MM (pain intensity: 0100 VAS)

    Oerlemans studyb 12 month follow-up (pain with effort) 135 13 4.5 [10.1 to 19.1]

    PT + MM vs OT + MM (pain: 078: McGill Pain Questionnaire)Oerlemans studyb 12 month follow-up 135 13 2.8 [1.4 to 4.2]

    PT + MM vs SW+ MM (pain: 078: McGill Pain Questionnaire)

    Oerlemans studyb 12 month follow-up 135 13 3.6 [3.2 to 4.0]*

    PT + MM vs OT + MM (volume difference between hands: mls)

    Oerlemans studyb 12 month follow-up 135 13 4.1 [6.1 to 2.1]

    PT + MM vs SW+ MM (volume difference between hands: mls)

    Oerlemans studyb 12 month follow-up 135 13 3.9 [2.2 to 5.6]

    PT + MM vs OT + MM (UL AROM: 05)

    Oerlemans studyb 12 month follow-up 135 13 0.3 [0.6 to 0]

    PT + MM vs SW + MM (UL AROM: 05)

    Oerlemans studyb 12 month follow-up 135 13 0.4 [0.1 to 0.7]

    Positive effect sizes favour a physiotherapy treatment effect.

    EMFT, electromagnetic field treatment; C, calcitonin; Ex, exercise; VAS, visual analogue scale; Rx, treatment; av, average; mls, milliliters; GMI, graded motor imagery; PT,

    physiotherapy; MM, medical management; NPS, neuropathic pain scale; mm, millimeters; NRS, numerical rating scale; OT, occupational therapy; SW, social work; UL, upper

    limb; AROM, active range of motion; Rec, laterality recognition; Im, imagined movements; Mir, mirror movements.a Estimated from a graph.b Effect sizes could only be calculated from data presented in Oerlemans (2000).* Statistically significant.

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    There is no evidence of sufficient quality to draw conclusions on

    the effectiveness of transcutaneous electrical nerve stimulation

    (Robaina et al., 1989) or stress loading exercise (Watson and Carl-

    son, 1987).

    There was insufficient primary evidence on the same interven-

    tion to allow grouping of results, which is a limitation of this re-

    view. Although every study on this topic may not have been

    sourced, this is unlikely, as the primary search strategies used eight

    databases as well as extensive secondary searching. The reviewers

    of the studies were not blind to author or source of the study and

    this may have affected the methodological quality rating. However,

    there was close agreement between the reviewers suggesting this

    is an unlikely source of bias.The majority of studies included in this systematic review

    (including all RCTs), investigated the effect of physiotherapy in

    combination with medical management. This means that while

    the added benefit of physiotherapy can be determined in a study

    that compares usual treatment with usual treatment plus physio-

    therapy (Durmus et al., 2004; McCabe et al., 2003; Moseley,

    2004, 2005, 2006), the stand-alone value of physiotherapy cannot

    be determined. In case series that involved usual medical manage-

    ment plus physiotherapy, (Pleger et al., 2005; Singh et al., 2004),

    the findings on physiotherapy effectiveness must be interpreted

    with caution as there was no comparison group and these studies

    were of lower methodological quality. Clinical guidelines support

    interdisciplinary management (Harden et al., 2006c; Stanton-Hicks

    et al., 2002) and the very nature of CRPS-1 requires that multiple

    disciplines contribute to the management of this painful, disabling

    and distressing condition. Therefore perhaps determining the ef-

    fect of physiotherapy in isolation is not a helpful exercise.

    4.2. Generalisability

    Consideration must be given to the generalisability of the stud-

    ies to current clinical practice, particularly with respect to partici-

    pant characteristics. Participants were recruited from University

    hospitals and specialist pain centres. These participants may have

    differing characteristics to other populations, such as severity and

    duration of symptoms, levels of depression and substance abuse

    and recalcitrance to primary management (Covington, 1996).

    Moseley excluded participants who had previously had a positiveresponse to sympathetic blockade (2004, 2005). Thus participants

    may have had sympathetically independent pain, which is associ-

    ated with a poorer outcome for CRPS-1.

    With the exception ofMoseley (2006), all RCTs recruited partic-

    ipants with only upper limb CRPS-1. Therefore their findings may

    not be generalisable to patients with lower limb CRPS-1. On the

    whole, participants were mostly female (66.8%), aged between 33

    and 53 years and had CRPS-1 symptoms in their upper limb for

    an average of 11.4 months following a noxious event, usually a

    fracture. These characteristics reflect epidemiological data on

    CRPS-1 (Janig and Stanton-Hicks, 1996; Low et al., 1996) but

    should be considered when generalising the results of this review.

    For example, the chronicity of participants in this review may inad-

    equately represent the effect some treatments may have on acute

    CRPS-1. Other reviews of mostly medical treatments for CRPS-1

    Table 8b

    Effect of Physiotherapy treatment on outcomes measured on a continuous scale, produced from other study designs

    Outcome measure (reference time of follow-up) Sample size Quality score Mean change between pre and post treatment (95% CI)

    Pain adapted sensorimotor treatment (pain: NRS 010)

    Pleger et al. (2005) end of Rxa (pain over the last 4 weeks) 6 9 3.8 [1.2 to 6.4]*

    Pain adapted sensorimotor treatment (% impaired tactile discrimination)

    Pleger et al. (2005) end of Rxa (2 point discrimination threshold) 6 9 23.0 [6.8 to 39.2]*

    Stress loading program (pain intensity: Verbal scale: 110 cm)

    Watson and Carlson (1987) 16 month follow-up 52 4 5.0 [N/A]

    Stress loading program (distance from finger pulp to palm crease: mm)

    Watson and Carlson (1987) 16 month follow-up 52 4 2.0 [N/A]

    Stress loading program (grip strength: kg)

    Watson and Carlson (1987) 16 month follow-up 52 4 16 [N/A]

    MVF+ usual Rx (pain intensity: VAS: 010)

    McCabe et al. (2003) end 6 weeks of Rx 8 10 4.1 [1.5 to 6.7]

    MVF+ usual Rx (temperature difference (painful vs non-painful)C)

    McCabe et al. (2003) end 6 weeks of Rx 8 10 0.9 [0.2 to 1.6]

    Graded exposure in vivo (TSK: 068)

    de Jong et al. (2005) end of Rx 8 12 34.5 [19.9 to 49.1]*

    de Jong et al. (2005) 6 month follow-up 8 12 35.0 [20.2 to 39.8]*

    Graded exposure in vivo (PHODA: 0100)

    de Jong et al. (2005) end of Rx 8 12 71.9 [68.3 to 75.5]*

    de Jong et al. (2005) 6 month follow-up 8 12 73.1 [69.9 to 76.3]*

    Rec (pain intensity: NPS: 0100)

    Moseley (2005) 20 12 8 [N/A]*

    Rec (function: NRS: 050)

    Moseley (2005) 20 12 7 [N/A]*

    Rec + Im (pain intensity: NPS: 0100)

    Moseley (2005) 20 12 16 [N/A]*

    Rec + Im (function: NRS: 050)

    Moseley (2005) 20 12 15 [N/A]*

    Im + Mir (pain intensity: NPS: 0100)

    Moseley (2005) 20 12 14 [N/A]*

    Im + Mir (function: NRS: 050)

    Moseley (2005) 20 12 19 [N/A]*

    Positive effect sizes favour a physiotherapy treatment effect.

    NRS, numeric rating scale; RX, treatment; %, percentage; N/A, not available; MM, mirror movements; TSK, Tampa Scale for Kinesiophobia; PHODA, Photograph series of Daily

    Activities; Rec, laterality recognition; Im, imagined movements; Mir, mirror movements; MVF, mirror visual feedback.a Lasting between 1 and 6 months.* Statistically significant.

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    have not reported participant characteristics (Forouzanfar et al.,

    2002; Kingery, 1997; Perez et al., 2001).

    4.3. Implications of variable diagnostic criteria

    It is essential to consider diagnostic criteria to ensure that the

    same condition is being evaluated in the studies contained in a sys-

    tematic review (Perez et al., 2007). Comparison of the diagnostic

    criteria reveals that important differences are present (Table 7).

    The studies by Watson and Carlson (1987) and Robaina et al.

    (1989)use basic signs and symptoms that are inadequate to diag-

    nose CRPS-1. On this basis alone, little weight can be placed on the

    results of their studies. Veldman criterion one (Veldman et al.,

    1993), has moderate sensitivity (0.65) and good specificity (0.85)

    (Perez et al., 2005). The IASP diagnostic criteria for CRPS-1 (Mers-

    key and Bogduk, 1994) have high sensitivity (0.98), but low speci-

    ficity (0.36) (Bruehl and Harden, 2001). These criteria have been

    revised to provide diagnostic criteria for research purposes (Bruehl

    et al., 1999). The proposed modified research diagnostic criteria for

    CRPS-1 have lower sensitivity (0.70) but higher specificity (0.96)

    (Baron and Janig, 2004).Moseley (2004, 2005, 2006) and de Jong

    et al. (2005)used the research diagnostic criteria in their studies.

    This suggests that they may have more accurately identified and

    excluded participants without CRPS-1 than those studies using

    the Veldman or IASP criteria.

    4.4. Implications of variable outcome measures

    The use of outcome measurement also requires consideration

    when interpreting the results of this systematic review. The com-

    parison of results between studies is limited due to the wide range

    of measurement domains and tools that were used (Table 6). Addi-

    tionally, the reliability and validity of outcome measures were

    rarely reported in the studies. This information is required so that

    the reader can determine the confidence they have in the measure-

    ment of the study variables and therefore the results of the study

    (Law et al., 1998b).Evidence of acceptable validity and reliability for neuropathic

    pain conditions such as CRPS-1exists forthe Neuropathic Pain Scale

    (Galer and Jensen, 1997; Jensen et al., 2005). The Impairment Level

    Sum Score is reliable and valid for upper limb CRPS-1 (Oerlemans

    et al., 1998) and lower limb CRPS-1 (Perez et al., 2003). The Rad-

    boud Skills Questionnaire has acceptable validity and reliability

    for upper limb CRPS-1 (Oerlemans et al., 2000a). The Walking Stairs

    Questionnaire and the Questionnaire Rising and Sitting Down are

    reliable for measuring activity limitation in lower limb CRPS-1 pa-

    tients (Perez et al., 2002). Greater credence can be attributed to the

    results of studies in which valid and reliable outcome measures for

    CRPS-1 were used (de Jong et al., 2005; Moseley, 2004, 2005, 2006;

    Oerlemans et al., 1999a,b, 2000b; Severens et al., 1999).

    4.5. Implications for further research

    Further studies would be strengthened by the use of the pro-

    posed modified research diagnostic criteria for CRPS-1 (Bruehl

    et al., 1999). The reporting of all CRPS-1 research would greatly

    benefit from international and interdisciplinary consensus on out-

    come measurement. This discussion should be informed by the

    WHOs International Classification of Functioning, Disability and

    Health (World Health Organisation, 2001). Critical appraisal and

    interpretation of future RCTS would be enhanced by adoption of

    the CONSORT statement checklist and flow diagram (Altman

    et al., 2001). However, while RCTs provide the highest levels of evi-

    dence, other forms of clinical investigation such as comparative

    studies and case series can be designed to provide clinically rele-vant evidence that can later be tested by RCT. The findings of this

    systematic review suggest that robust RCTs should be designed to

    test the treatments for which there was good quality evidence

    from case series, i.e. graded exposure in vivo (de Jong et al.,

    2005) and interdisciplinary outpatient treatment programs (Singh

    et al., 2004).

    4.6. Implications for clinical guidelines

    Existing CRPS clinical guidelines (Harden et al., 2006c;Stanton-

    Hicks et al., 2002) should be interpreted with caution, as the rec-

    ommendations for specific physiotherapy treatments are not based

    on current evidence. The findings of this review would support the

    inclusion of graded motor imagery (Moseley, 2004, 2005, 2006) for

    which there is good to very good quality level II evidence from

    three RCTs produced by one research group. These results need

    to be replicated by others. Stress loading exercise (Watson and

    Carlson, 1987), which is included in clinical guidelines on CRPS

    (Harden et al., 2006c; Stanton-Hicks et al., 2002) has very little evi-

    dence supporting its effectiveness. We recommend that clinical

    guidelines should be evidence based.

    The findings of this systematic review can be used to update

    recommendations for the physiotherapy management of CRPS-1

    to ensure these recommendations are clinically effective, relevant

    and based on the best available evidence; to stimulate interdisci-

    plinary discussion about physiotherapy and to inform future

    research.

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