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Mirror box therapy added to cognitive behavioural therapy inthree chronic complex regional pain syndrome type I patients:a pilot studyY.I.G. Vladimir Tichelaara, Jan H.B. Geertzenb,c, Doeke Keizerd andC. Paul van Wilgenb,d Complex regional pain syndrome type I is a disorder syndrome type I may facilitate rehabilitation. Measuring of the extremities with disability and pain as the most whether the affected limb still belongs in the patient’s prominent features. This paper describes the results of body scheme could be of prognostic value in the cognitive behavioural therapy combined with mirror box treatment of chronic complex regional pain syndrome therapy in three patients with chronic complex regional type I patients. International Journal of Rehabilitation pain syndrome type I. Before, during and at follow-up the following measurements were assessed: pain (visual analogue scale, 0–100), range of motion, musclestrength, and the areas of allodynia and of hyperalgesia.
International Journal of Rehabilitation Research 2007, 30:181–188 Furthermore, patients were asked for their feelingsand thoughts about mirror box therapy and about the Keywords: cognitive behavioural therapy, complex regional pain syndrometype I, mirror box therapy, rehabilitation, chronic affected limb. Pain at rest, pain after measuring allodynia/hyperalgesia and pain after measuring strength decreased.
aUniversity Medical Centre Groningen, University of Groningen, Range of motion improved in two patients. Strength bCentre for Rehabilitation, cNorthern Centre for Health Care Research and dDepartment of Anaesthesiology, Pain Centre, University Medical Centre improved in one patient. The area of hyperalgesia Groningen, University of Groningen, The Netherlands increased for all three patients, whereas the areaof allodynia remained stable in two patients and Correspondence to Prof Jan H.B. Geertzen, MD,PhD, Centre for Rehabilitation, decreased in one patient. Two patients felt that their University Medical Centre Groningen, University of Groningen, Groningen,The Netherlands affected limb still belonged to them, one did not.
Tel: + 31 503612295; e-mail: [email protected] Cognitive behavioural therapy combined with mirror boxtherapy for patients with chronic complex regional pain Received 19 October 2006 Accepted 29 January 2007 resulting in disuse and a painful, dystrophic or atrophic, Complex regional pain syndrome type I (CRPS-I) is a dysfunctional limb (Veldman et al., 1993). Allodynia is disorder of the extremities with disability and pain as the defined as pain due to a stimulus which does not normally most prominent features, especially in chronic CRPS-I provoke pain (Mersky and Bogduk, 1994). Hyperalgesia is patients (Ribbers et al., 1995, Geertzen et al., 1990).
defined as an increased response to a stimulus which is CRPS-I is defined by diagnostic criteria proposed by the normally painful (Mersky and Bogduk, 1994).
International Association for the Study of Pain. In CRPS-Ithere is no evidence of nerve damage, in contrast to Several theories are available, which may account for signs CRPS-II (causalgia) (Stanton-Hicks et al., 1995). Pain is and symptoms in the chronic phase of CRPS-I. In the usually located in the distal part of the limb, and has a learned-nonuse theory, peripheral and central sensitiza- tendency to spread proximally (Rommel et al., 1999).
tion will lead to allodynia or hyperalgesia in CRPS-I.
Spreading of signs and symptoms beyond the site of Immobility and disuse occur as a result of formerly initial trauma is characteristic of CRPS-I (Veldman, received negative feedback (pain or failure) when trying 1995). Women are more frequently affected than men to use the affected limb (Woolf et al., 1994; Schu et al., 1999). As a consequence, when disuse of the limbremains for a longer period of time this may lead to more In the acute phase, the five classical symptoms of atrophic changes, immobility and cortical reorganization inflammation (tumor, rubor, kalor, dolor and functio of the somatosensory cortex (Bortz, 1984).
laesa) may all be present (Veldman, 1995). In the chronicphase of the syndrome (i.e. with features of CRPS-I for 6 In the remapping hypothesis, in patients with chronic months or longer), pain, sensory changes (allodynia and CRPS-I, absence of consistent proprioceptive feedback hyperalgesia) and trophic changes are more prominent, when giving motor commands to the affected limb may Copyright Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
International Journal of Rehabilitation Research increase pain and changes in the primary somatosensory cases of patients with chronic CRPS-I, treated with CBT cortex in patients (Ramachandran and Hirstein, 1998).
Evidence of underlying changes in the primary somato-sensory cortex was found in patients with CRPS-I using a magneto encephalogram or functional magnetic reso- CBT consists of the following phases. In the preclinical nance imaging (Maihofner et al., 2003; McCabe et al., phase, after multidisciplinary assessment, reconceptuali- zation of the patients’ cognitions about CRPS-I isestablished. The so-called sensitization model is used Recently, studies have described that one could possibly to explain signs and symptoms to the patients (van break through the vicious circle of pain and disuse as a Wilgen and Keizer, 2004). The main goal of this result of remapping the primary somatosensory cortex by reconceptualization of cognitions is to convince patients providing visual feedback (Maihofner et al., 2003, 2004).
to no longer believe that actual tissue damage is As described in patients with phantom pain and responsible for their pain and dysfunction. In the clinical sensations, providing visual feedback as a substitute for phase, operant, cognitive and respondent techniques are missing proprioceptive feedback may reduce pain, used by an experienced team consisting of a psychologist, enabling patients to experience a more ‘vivid’ phantom physical therapist and a physician. A time-contingent (Ramachandran, 2000). Also a central role for the detoxification protocol is implemented during the first premotor cortex could be present. When normal somato- week. After detoxification, mirror box therapy is intro- sensory feedback is missing, visual feedback restores the duced during the second week, as add-on to the information flow from the posterior parietal cortex to the premotor cortex (Di Pelligrino et al., 1992; Seitz et al.,1998; Altschuler et al., 1999). Recruiting the premotor Three patients participated in a 4–6 weeks inpatient cortex or rebuilding the motor programme in the CBT combined with mirror box therapy aiming at premotor cortex by providing visual feedback could regaining limb function and pain reduction. During the reduce pain and facilitate the limb movement (Rothgan- first week, all analgesics were gradually reduced or stopped (detoxification), as discussed with the patientin the preclinical phase. In the second week, mirror To achieve visual feedback, patients can be treated with therapy was introduced three times a day for two cycles of mirror box therapy, in which their limbs are positioned in 5 min. Patients exercised little movements of the a box separated by a mirror placed saggitally. By looking in nonaffected side, whereas they were instructed to the mirror at the unaffected side, patients can be ‘fooled’ imagine the movement was performed in both limbs.
in believing that the affected limb is moving effortlessly During this procedure patients looked at their unaffected (Ramachandran and Hirstein, 1998). In patients with limb in the mirror, so that it would appear as if both limbs hemiparesis after stroke, mirror therapy has been used in were moving effortlessly. When the patient was able to providing visual feedback to reduce pain and facilitate perform little movements with the affected limb (with rehabilitation of the affected limb [Altschuler et al., 1999, the toes) he or she was encouraged to exercise these Rothgangel et al., 2004]. Also in patients with phantom movements with both limbs while looking in the mirror.
limbs and phantom pain, mirror box therapy has been In the third week mirror box therapy was performed five used successfully (Ramachandran, 2000).
times a day, for two cycles of 5 min.
Concordantly, mirror box therapy in patients with CRPS-I Measurements were performed by an investigator who existing for less than 2 years has shown to cause some was not involved in the treatment. The patients were regain of functionality and mobility, and to reduce pain evaluated before the clinical phase, once a week during (McCabe et al., 2003a). Evidence of cortical reorganiza- therapy (the mean scores are presented as one) and at tion of the primary somatosensory cortex was also found follow-up after the clinical phase. The first patient was in parallel with clinical improvement of the patients evaluated at 14 weeks follow-up, the second at 8 weeks (Maihofner et al., 2004). In our hospital, patients with follow-up and the third at 5 weeks follow-up.
CRPS-I are treated with cognitive behavioural therapy(CBT).
Quantitative aspects of pain were assessed using a visualanalogue scale (VAS, range 0–100). The pain was Owing to the formerly mentioned results with mirror box measured at rest, and after testing range of motion therapy, we decided to add mirror box therapy to CBT in (ROM), muscle strength, allodynia and hyperalgesia.
the treatment of three CRPS-I patients in our hospital.
We tried to measure some outcomes to establish an idea ROM was measured using a goniometer, to assess whether mirror box therapy could be a useful add-on to maximal hand (dorsal/palmar flexion) or feet (dorsal/ CBT in the treatment of CRPS-I. We will describe three plantar flexion) movements. In addition, the position of Copyright Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Mirror box therapy added to cognitive behavioural therapy Tichelaar et al.
the hand or foot in which the patient was most Result of case 1 before, during and after CBT with mirror comfortable at rest was recorded. Muscle strength of the affected limb was measured with a hand-held dynamometer according to a standardized protocol using break tests (van Wilgen et al., 2003). Allodynia and hyperalgesia were assessed using a brush and a Von Frey monofilament, respectively. With the brush Ab-fibre- mediated allodynia was tested, whereas with the Von Frey monofilament (no. 4.98) Ad-fibre nociceptors and thus hyperalgesia were tested. The upper borders of the areas with allodynia and hyperalgesia were measured. From each digit of the affected limb, a virtual line was extended proximally, passing an anatomical landmark CBT, cognitive behavioural therapy; VAS, visual analogue scale; ROM, range of (for the ankle, the malleoli; for the wrist, the styloid- proces of the radius), which was chosen as zero-point.
The brush or Von Frey monofilament moved distally froman area where no allodynia or hyperalgesia was present, described it to be more like a funny feeling. At the end of towards the affected area, along the five virtual lines of the treatment, the patient’s foot felt like it was moving, the five digits. When the patient perceived the stimulus but he did not see it moving in reality.
to be painful, the distance between the anatomical zero-point and the judged painful stimulus, along each virtual At follow-up the patient was able to walk very slowly, line, was measured. The brush moved continuously at a for little distances without using his elbow-crutches.
speed of approximately 2 cm/s and the monofilament was He also stated that mirror box therapy improved his pressed on the skin for 1 s, with intervals of 0.5 cm. At condition and experienced less pain without using investigation, first the brush and thereafter the Von Frey VAS scores, results of ROM and of strength tests are Finally, patients were asked to write down their thoughts listed in Table 1. The course of the areas of allodynia and about mirror box therapy and about their affected limb, hyperalgesia is shown in Figs 1 and 2, respectively.
before, during and after CBT combined with mirror box Overall, pain decreased. ROM (dorsal flexion) increased and the position of the foot at rest turned from 45towards 51 plantar flexion. This pes equinus restricted further progress of mobility; reconstruction surgery is currently considered. Strength improved, the area of This patient was a 23-year-old man, who developed allodynia decreased, but the area of hyperalgesia CRPS-I after a fracture of digit III in his right foot, 30 remained almost stable (Figs 1 and 2).
months before attending our hospital. Treatment withphysical therapy, transcutaneous electrical nerve stimula- tion, a sympathetic block and medication did not improve This patient was a 42-year-old woman with CRPS-I of the left leg as a result of a minor trauma to the left knee 8months earlier. For this condition she received pharma- Patient used two elbow-crutches for walking, carefully cotherapy and physical therapy. Pain was always there and avoiding using the affected foot. He was not able to move was described as burning, descending from the left knee his foot and did not exercise or touch his foot at all. Pain distal towards the toes. The patient sat in a wheelchair, was always present. At the time of multidisciplinary and was unable to walk. The leg had a bluish colour, assessment, the right foot was oedematous, allodynic and mostly distal. The knee was in 201 flexion position, with fixed in 451 plantar flexion at rest. Increased hair and nail atrophic changes of the quadriceps muscle. The whole growth was seen (Fig. A1 of the Appendix). In the leg appeared sweaty, hyperpathic and allodynic. Hair preclinical stage he used vitamin C, nifedipine and growth was not visible on the distal part of the leg; there was a complete nonuse of the left leg (Fig. A2 of theAppendix). Daily medication at intake was tramadol, Before mirror box therapy, the patient described his celecoxib and amitriptyline. Medication after detoxifica- bodily sensations of his leg as if it was not responding, although he commanded it to move. During mirror boxtherapy, he initially reported an incongruent feeling She described the affected leg as still belonging to her, seeing the affected foot moving in the mirror. Later, he but like it was not willing to move. Besides that, when Copyright Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
International Journal of Rehabilitation Research Pain area of Ab (allodynia) of case 1. Before treatment (Á-Á-Á-Á-Á), duringtreatment ( – ) and follow-up (- - - -).
Pain area of Ad (hyperalgesia) of case 1. Before treatment (Á-Á-Á-Á-Á),during treatment ( – ) and follow-up (- - - -).
trying to move or touching the leg, she experienced a lotof pain.
of the foot at rest worsened a little, it was held more inplantar flexion at follow-up than before treatment. The During mirror box therapy, she indicated repeatedly it felt area of allodynia decreased but the area of hyperalgesia like the leg was not responding to her commands. She never experienced the feeling of movement of the leg, anddid not see it moving at all. After mirror box therapy, she was disappointed not making contact with the limb.
The third patient was a 46-year-old woman; 9 years agoshe was involved in a car accident, which resulted in the At follow-up, the situation did improve a little, i.e. the development of CRPS-I in her left shoulder, nondomi- patient used less medication and experienced less pain, nant arm and hand. Physical therapy and sympathetic but was still not able to move the affected leg. The blocks did not improve complaints. At the time of patient was disappointed by the results. It has to be multidisciplinary assessment, flexion contractures in noted that during therapy, she experienced a major life shoulder, elbow, wrist and fingers were present. Except event, which decreased her motivation and interrupted little movements of the thumb and digit II, extension of the fingers was neither passively nor actively possible.
The forearm was cold and atrophic (Fig. A3 of the Table 2 shows the VAS scores and the result of ROM Appendix). Pain was not always present; mostly it was tests. Measuring strength was not possible, because the provoked by trying to move the arm, or by contact with pressure of the hand-held dynamometer caused too much surroundings or cloth. Allodynia and hyperalgesia were pain. In Figs 3 and 4 the course of the areas of allodynia present. She was not using any medication at intake.
and hyperalgesia is shown. Pain at rest and after testingallodynia and hyperalgesia decreased. Dorsal flexion Before mirror box therapy she stated her arm did not increased a little, but plantar flexion decreased. Position belong to her anymore. She felt it was like something Copyright Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Mirror box therapy added to cognitive behavioural therapy Tichelaar et al.
Result of case 2 before, during and after CBT with mirror CBT, cognitive behavioural therapy; VAS, visual analogue scale; ROM, range ofmotion.
Pain area of Ad (hyperalgesia) of case 2. Before treatment (Á-Á-Á-Á-Á),during treatment ( – ) and follow-up (- - - -).
Result of case 3 before, during and after CBT with mirror CBT, cognitive behavioural therapy; VAS, visual analogue scale.
Pain area of Ab (allodynia) of case 2. Before treatment (Á-Á-Á-Á-Á), during In Table 3 the results of VAS are shown. ROM and treatment ( – ) and follow-up (- - - -).
strength testing were not possible because of severedystonia and contractures of the affected arm. The courseof the area of allodynia and hyperalgesia is shown in Figs 5 strange to her, she even dreamed of herself without and 6. Pain at rest and after testing allodynia and having a left arm. The patient was not even able to hyperalgesia decreased during treatment, but at follow- imagine her hand moving as it could before the accident.
up she still experienced pain attacks.
During mirror box therapy the patient did not recognize In this small group of CRPS-I patients with severe disuse the affected left arm as belonging to her. She also did not and pain, mirror box therapy was added to CBT as experience any feelings of making contact with her arm.
After treatment, the patient did not improve on anyoutcome. She still could not move the arm and the pain After treatment and follow-up we can conclude that case attacks remained happening on movement or touching of 1 improved, i.e. he experienced less pain without using any medication and could walk a little distance without Copyright Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
International Journal of Rehabilitation Research Pain area of Ab (allodynia) of case 3. Before treatment (Á-Á-Á-Á-Á), during Pain area of Ad (hyperalgesia) of case 3. Before treatment (Á-Á-Á-Á-Á), treatment (—) and follow-up (- - - -).
during treatment (—) and follow-up (- - - -).
using elbow-crutches. The patient in case 2 improved resulting from the duration of the CRPS-I. In addition, less. She experienced less pain, but mobility did not our second patient improved at least on pain. So chronic improve. In the last case, the patient did not improved CRPS-I may not be susceptible to CBT and mirror box therapy. Whether this is caused by peripheral pathology(contractures, atrophy) alone or also by irreversible Besides these results, it seems that the outcome of our cortical changes of the primary somatosensory cortex combined treatment for CPRS-I could be predicted by some factors identified in these three case studies.
In addition, patients stating that their affected limb does First, as mentioned earlier, the longer the CPRS-I not belong to them anymore (i.e. is not a part of their diagnosed, the worse the disability and pain (Veldman body scheme anymore), seem to have no benefit of CBT et al., 1993). In addition, mirror therapy alone does not combined with mirror box therapy (case 3). In a lesser seem to improve disability when CRPS-I exists longer way, patients saying they cannot imagine or feel their than 2 years (Bortz, 1984). In our third case, CRPS-I was affected limb moving in their mind, i.e. patients who are existing for almost 9 years, which had led to irreversible not able to make contact with their limb, also seem to contractures and atrophy. Although in this case it may have less benefit of our treatment (case 2). Alltogether, seem too obvious that such a subtle approach as CBT and some chronic CRPS-I patients might have benefit from mirror box therapy can do little about this major CBT and mirror box therapy, in making ‘contact’ with the irreversible pathology, in other cases where CRPS-I does not exist that long, it may be less clear. In our first case, McCabe et al., 2003a; Rothgangel et al., 2004). An where CRPS-I existed for 2.5 years, the patient did important prognostic value then might be the degree of improve, but this was also limited by contractures ‘foreignness’ of the affected limb described by patients.
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Mirror box therapy added to cognitive behavioural therapy Tichelaar et al.
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International Journal of Rehabilitation Research The results found in cases 1–3 are shown in Figs A1–A3.
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