Abstract
Complex Regional Pain Syndrome is pain syndrome confined to one or more limbs, usually occurring after trauma. The cause of CRPS remains unknown and its diagnosis is based upon a set of clinical signs and symptoms: the Budapest Criteria (figure 1). CRPS is characterised by the presence of limb pain associated with sensory, motor, autonomic, skin and bone changes. It is often resistant to treatment and its clinic course is difficult to predict. Early diagnosis and treatment from a multidisciplinary team is associated with the best outcome. In the UK there has been recent guidelines published to aid the diagnosis and treatment of CRPS. The mainstay of this treatment is physiotherapy, patient education and the medical management of pain. The patient in this case was scheduled to undergo an amputation of the affected limb prior to trying any of the above management, indeed prior to even being diagnosed with CRPS. Amputation for the treatment of CRPS is controversial. Evidence based guidelines regarding CRPS currently state that there is insufficient evidence to prove that amputation positively contributes to the treatment of the patient. It also runs the risk of the patient suffering from phantom limb pain or CRPS recurring in the stump or contralateral limb. If indeed CRPS is a sympathetically mediated neuropathic pain, as proposed, surgery to the area is likely to aggravate the condition. Tissue damage locally will result in the release of inflammatory neurokines leading to up regulation of the immune and nervous system. Amputation in the presence of CRPS can only therefore be justified in the treatment of therapy-resistant infection after other treatment options have been explored. Even more pertinent to this case was that the patient had never tried more ‘conventional’ treatment options before an amputation was decided upon.
Case Presentation
Referral Information
Patient JM was referred by Dr FJ, consultant in rehabilitation medicine as a ‘query’ diagnosis of CRPS of his right lower limb. JM was scheduled to have a Symmes amputation, under a Professor MS a consultant orthopaedic surgeon, two weeks after this referral was made.
Questionaire Scores
MPSQ 8
Mod Zung 21
Current Problem
JM complained of gradually worsening pain in right ankle and foot over a twenty-four month period. He scored his pain from 2-8/10 worse on light touch, cold weather and at the end of the day. The pain was accompanied by decreased motor function, oedema, an increase in sweating and skin colour changes in the limb. He described his pain as like a really bad ‘tooth ache’ especially worse when the limb was swollen and only decreased by rest and elevation. He described a ‘dropping sensation’ in the foot despite it being fused at the ankle which causes such severe pain it has at times caused him to vomit.
Background History
JM was born with a congenitally abnormal right tibia. As a child he underwent multiple operations on his right ankle. He has had two osteotomies, a bone graft and Lizorov frame and a triple arthrodesis at this ankle joint.
Past Medical History
JM has no other medical problems.
Drug History
Dihydrocodine 30mg four times a day. No other medication tried.
Family History
No family medical history of note. Mum and Dad alive and well.
Social History
JM lives with his parents and works in IT full time from an office at home. He feels that his career progression has been halted over the last few years as his pain has increased and his function worsened. He has an active social life and close family support. He played regular tennis until the pain in his ankle increased a few years ago but hasn’t been able to play since 2010. He denies any depression or anxiety although finds his functional limitations frustrating. His mother attended his first clinic appointment and was extremely upset and anxious when it was suggested that her son try medical treatment options before resorting to a Symmes amputation. She felt that JM had gone through a lot of psychological distress coming to terms with the prospect of having his leg amputated at the foot and was fully decided this was the best course of action only for that decision to be questioned. JM didn’t have any expectations of the pain clinic other than to help him reduce his pain to a ‘livable level’. The main reasons given by the Orthopeadic team for amputation was functional: to improve his gait and in the long run reduce possible arthritic changes developing due to his poor posture.
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Examination Findings
JM’s right lower limb on inspection was swollen from below the knee, with a pale discolouration and multiple operation scars at the ankle. There were noticeable trophic changes in the nails of the right foot but no hair or skin changes locally. His right ankle is fused in fixed dorsi flexion. On palpation it is cooler to touch and clammy when compared to the left lower leg and ankle. JM is tender to light touch over the medial aspect of the right ankle. The right knee has normal and has a good range of movement.
Diagnostic Formulation
JM’s right ankle is positive for the Budapest Diagnostic Criteria for Complex Regional Pain Syndrome.
Sensory
Allodynia
Hyperalgesia
Vasomotor
Temperature asymmetry
Skin colour changes
Skin colour asymmetry
Sudomoter / Oedema
Oedema
Sweating changes
Sweating asymmetry
Motor / Trophic
Decreased range of movement
Motor dysfunction
Trophic changes (hair/skin/nails)
For the patient to be positive for CRPS with the Budapest criteria they must have continuing pain which is disproportionate to the eliciting event and have:
More than one sign in two or more of the categories above AND
More than one symptom in three or more of the categories above AND
No other diagnosis that could better explain their signs and symptoms.
JM has all of the italic signs and symptoms above and no other diagnosis that could better explain his symptoms. JM had also undergone a bone density scan of the right leg showing osteopenia and an xray showing arthritic changes to the deformed and fused ankle joint. Therefore a diagnosis of CRPS of his right lower limb was made.
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Subsequent Management
JM was started on lignocaine 5% patches. Communications were also made with the other consultants involved in JMs care (orthopaedics and rehabilitation medicine). The orthopaedic team were keen to point out that the benefits of the surgery were functional and that even if JM were to become pain free with conservative treatment then it would not solve his functional problems. At follow up one month after starting the lignocaine 5 % patches JM was managing extremely well. His pain was down to a manageable level and by wearing the patches he could function nearly normally. He is now walking without a stick and had managed to return to playing tennis and had attended a work conference. He had decided against having the amputation and was happy to carry on with the current medical management of his condition. Further treatment options, should his pain flair up again, were discussed. These included IVRA (intravenous regional anaesthesia) and a spinal cord stimulator.
Discussion
CRPS is a pain syndrome with an unclear pathophysiology and unpredictable clinical course. The diagnosis of CRPS is based upon a set of signs and symptoms derived from the history and examination of the patient. The treatment of CRPS is aimed at improving function and requires the use of a interdisciplinary team encompassing physiotherapy, psychological therapies and pain management. The management of CRPS depends on prompt diagnosis and early management as response to treatment is adversely affected by any delays. In the UK recent guidelines have been developed for the diagnosis and management in the context of both primary and secondary care (2). JM in this case had been managed primarily by an orthopaedic team who had not linked his symptoms with a diagnosis of CRPS. CRPSs management requires a multidisciplinary approach based upon the published ‘Four Pillars of Intervention’ (3).
The Four Pillars of Treatment for CRPS
Patient information and education
Patients should be provided with appropriate education about CRPS to support self management
Patients should be reassured the physical and occupational therepy are safe and appropriate and engaged in the process of goal setting and review
Pain relief (medication and procedure)
No individual drug can be recommended at current time due to lack of evidence but the following may be considered
a)neuropathic pain medication
b)pamidronate 60mg iv single dose in pts with CRPS <6 months
c)spinal cord stimulator
Physical and vocational rehabilitation
Emphasis on restoration of normal function with techniques such as pacing, graded exercise
May also include techniques such as self administered desensitization, graded motor imagery, mirror feedback, mental visualisation
Psychological intervention
Based on an individual assessment to identify and manage any factors which are perpetrating pain and/or disability. Treatments including CBT, family support.
The interesting question posed by this case was whether to amputate a limb in the presence of CRPS. A review article in the Journal of Bone and Joint Surgery was published in October 2011 (4). In this review they looked at all the published articles looking at amputation as a treatment for CRPS. They concluded that there was insufficient evidence to support such treatment (of the 161 articles they identified there were only 26 studies and none of them represented higher than level 4 evidence). None of the evidence they found could clearly delineate the benefits and adverse effects of an amputation performed in the presence of CRPS. This team were also looking at ‘Therapy-resistant CRPS’. JM in this case study had not formally been diagnosed with CRPS and had never tried any of the conventional non surgical treatment options. To proceed straight to amputation which in itself can lead to significant deafferentation pain without first trying more conservative options would seem foolhardy. Amputation in the presence of CRPS can lead to worsening of the pain and has led to CRPS moving post amputation into another of the patient’s limbs. The study concluded that the only evidence in favour for amputation was in the presence of severe concurrent infection in the limb resistant to conventional treatment options (antibiotics +/- minor surgery). Of the studies amputation was carried out for pain in 80% and dysfunction in 72%. However 31/65 patients had recurrence of CRPS in their stumps and 11/65 went on to suffer from phantom limb pain.
Lignocaine 5% patches are currently only licenced for the treatment of post herpetic neuralgia. It is however regularly used ‘off licence’ for many causes of chronic pain especially neuropathic pain. In the BJA in 2007 (5) a case study was published sighting a patient with CRPS in his right hand who responded very well to the use of lignociane 5% patches. This patient showed an 80% improvement in his pain and movement within two weeks of starting the treatment and had previously shown no improvement with conventional treatments (physiotherapy, neuropathic pain medication, stellate ganglion blockade). A pilot study was carried out in patients with CRPS using 5 % lignocaine patches in 2000 by Devers and his colleagues. They reported moderate pain relief in 81% of the participants. The safety, pharmacokinetics and tolerability of lignocaine 5% patches were studied in 2002 by Arnold and his colleagues (6). They failed to produce a plasma lignocaine concentration above that needed to produce a toxic effect despite applying four plasters 12 hourly. It is therefore a very safe treatment when used within its prescribed limits (no more than three patches at a time for twelve hours followed by a twelve hour break). Lignocaine 5% are an unlicensed but possible treatment option in CRPS. The main problem with the patches are their cost (thirty patches cost £72.40 (7)). However in JM’s case the use of the lignocaine patches are currently enabling him to avoid an amputation operation and participate as an economically productive member of society therefore from a health economics point of view are beneficial to all concerned.
Future treatment options for JM included a spinal cord stimulator (SCS). A SCS
Conclusions
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