Joopm Vol 1 Ed 3 (2014)

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Vol 1, No 3 (2014)

Courtesy of the Robert Pope Foundation

An open access, online journal covering all aspects of pain

ISSN 2047-0800


Vol 1, No 3 (2014) April 2014

Editor-in-Chief Dr Rajesh Munglani Pain Medicine Consultant

The Journal has been created in recognition of the fact that not all information or advances in pain medicine can be presented in a randomised controlled fashion.

The editor welcomes contributions. Please email kate@joopm.com to submit an article for consideration.

The Journal will publish papers on clinical practice, basic science, ethics and medico-legal aspects of pain. Issues around suffering, theological, social, psychiatric, psychological, education and resources limitations in pain medicine will also be considered.

www.joopm.com email: info@joopm.com Editorial Board Assistant Editor-in Chief Dr Turo Juhani Nurmikko

Contents Interventions for cervical joint facet pain Shamim Haider

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Cervical meningioma in a patient with lumbar pain: a case report Alan Fayaz, Serge Nikolic

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Numbers needed to heal, numbers needed to harm, numbers needed to kill; reflections on opioid therapy and primary duty of medicine Rajesh Munglani

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Current understanding of the causes of Complex Regional Pain Syndrome (CRPS), with its Medico-Legal Implications Andreas Goebel

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Stent- mediated redistribution of cerebral venous outflow in the treatment of severe intractable headache Nicholas Higgins, Frances Hall, John Pickard

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CRPS Case Study: return to work rehabilitation using graded motor imagery, exercise and work hardening Deborrah Kim Thornhill

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Early Treatment in CRPS: A Patient’s Perspective Zoe Branka Holland

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Clear Effect of Mirror Therapy on Trigeminal Neuralgia Pain Annegret Hegenberg

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Estimating the Prevalence of Chronic Pain in a Given Geographical Area Martin Seymour, Stephen Patterson

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Pain Fellow Representative Dr Kiran Sachane

Consent Andrew Lawson

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Copy Editors Paul Nash Louise Mantin

‘Experts are forever’ or ‘ You only live once?’ A Cautionary Tale of Duty and Temptation in Compensation Litigation Julian Benson

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The impact of the Jackson reforms on medico-legal evidence Harriet Formby

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Psycho-Legal Assessment of Chronic Pain and Symptom Validity: A UK Perspective Vicki Hall

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Should the Law decide on moral issues? Robert Walker

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Assistant Editors Dr Joshua Adedokun Dr Ganesan Baranidharan Dr Arun Kumar Bhaskar Revd Dr Mark Quinn Bratton Rev Canon Dr Adrian Francis Chatfield Dr Neil Collighan Dr Andrew Cooper Dr Simon Dolin Mr Marcus Grant Dr Sanjeeva Gupta Dr Shamim Haider Dr Dalvina E. Hanu-Cernat Dr Kevin Markham Dr Patrick McGowan Dr Vivek Mehta Dr Charles Pither Dr Andrew Ravenscroft Dr Jonathan Richardson Dr Manohar Lal Sharma Dr Michael D. Spencer Dr Simon James Thomson Mr Michael Walsh

Journal Manager Ms Kate Dougherty KJD Communications www.kjdcommunications.com

ISSN 2047-0800


Interventions for Cervical Facet-Joint Pain Dr Shamim Haider MBBS, MD, FRCA, FFPMRCA Consultant (pain medicine), Colchester Hospital University NHS Foundation Trust

Abstract This is a case discussion of a young man with bilateral neck pain, which responded well to Median Branch Blocks, but no added benefits with subsequent radiofrequency (RF) denervation. Several variations of RF technique including cooled RF, positioning, as well as bipolar approach, were suggested. Trials of botox for muscle spasm and facet-joint injections have been proposed. Cervical epidural injection is suggested as the next step, with a following discussion on various techniques employed by the clinicians. Keywords Chronic, neck, cervical, pain, facet joint, median branch blocks, radio-frequency, cooled RF, bipolar, botox, cervical epidural, particulate steroid

Introduction Neck pain is the second most common presentation at the pain clinics and facet-joint is implicated in large proportion of these patients. As part of a multi-modal approach, Median Branch Blocks (MBBs) have a specific role in well-selected patients, with good response-rate. Among responders, radio-frequency (RF) denervation is usually the next logical step to maximise the therapeutic benefits of MBB. Although evidence for cervical RF is fair in providing lasting and complete relief, it is well recognised that RF neurotomy works only in a moderate proportion of patients. There are various reasons for failure, including initial placebo response to MBB, improper patient selection, technical failure and unmasking of other pain symptoms.1 Further available interventional options should be considered after careful revaluation of the reasons for failure.

Problem A young professional male patient with thick ‘bull’ neck presented with bilateral neck pain. The patient could not tolerate large doses of analgesics and bilateral MBBs gave excellent pain relief. Subsequently, RF denervation was attempted, which failed to produce any added benefit compared with MBB. The denervation was carried out using unipolar RF 20 G: 10 cm needle with a 10 mm active tip. The procedure was done at C3–6 levels bilaterally in lateral position. There were technical difficulties with the procedure as proper electrode placement was difficult due to the large size of the patient’s neck. Additionally, the lower cervical region beyond C6 was not well visualised on fluoroscopy; hence, denervation could not be attempted in this area.

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Recommended treatment plans Intra-articular injection A trial of intra-articular injection of steroids may be helpful when history and response to MBB are typical of cervical facet-joint pain

Variations of RF technique Prone position is regarded to offer the best conditions for cervical medial branch RF in patients with a thick neck. An alternative suggestion is to use the lateral position with slight extension of the neck. A bipolar RF approach of putting needles at the top and bottom of the lamina would create a larger strip lesion, which would have a higher chance of catching the medial branch. However, newer RF machines would be needed for this approach. Cooled RF is a relatively new technique for neurotomy in the cervical region, which is claimed to deliver larger volume lesions compared with conventional RF. There is hardly any published data on cooled RF in the cervical region. A retrospective study for management of sacro-illiac joint pain found that the use of cooled RF (in 20 patients), seemed to be associated with a higher percentage of positive outcomes.2 On the other hand, a recent study found little evidence of a significant relationship between the two RF techniques (conventional and cooled) and duration of pain relief in management of sacro-illiac pain.3

Botox The persistence of muscle spasm in the posterior compartment is suggested as one of the reasons for non-response to RF, when there was a good response to MBB. Injection of botox with fluoroscopic guidance, while performing RF denervation, could potentially overcome this. However, a Cochrane review in 2011 concluded that the available evidence does not support the use of botox either as a monotherapy or in combination with any other treatment in patients with subacute or chronic neck pain.4

Cervical epidural Trans-laminar cervical epidural injection of local anaesthetic and steroid is regarded as the next option, especially for bilateral widespread neck pain. However, there is very little published evidence of its long-term efficacy. Technique for cervical epidural The inter-space selected for trans-laminar epidural is almost always at C7–T1 or below, as the epidural space above this level is very narrow. T1–2 level is preferred by some because the ligamentum flavum is tougher at this level. However, there is about a one-in-five chance of a gap in the ligamentum, even at this level, making mid-line loss-of-resistance technique challenging. Hence, the technique of going from side, walking-off lamina and use of dye to confirm epidural space is safer, especially in inexperienced hands. To reach the higher cervical levels, use of an epidural catheter passed up to C3 has been suggested. This technique involves injection of 5 mls of local anaesthetic (LA) and steroid mixture, while slowly withdrawing the catheter. 4

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Both prone and sitting positions are employed for the epidural. In the sitting position, it is easy to get shoulders out of the way, especially in those with a thick neck or unusual anatomy. However, episodes of fainting are not uncommon with sitting. 18 G or paediatric touhey needles are used by many. Some operators practise use of 16 G, as it provides good resistance and hence good loss of resistance. Hanging-drop technique, loss of resistance, as well as walking-off lamina under fluoroscopy is used to identify the epidural space. However, the classical pattern of spread of dye in the epidural space on X-ray is the only sure way to confirm it. There is some evidence to indicate that use of particulate steroids provides slightly longer pain relief compared with non-particulate ones. However, there is a consistent move among practitioners, away from use of particulate steroids anywhere in the cervical region, due to case reports of ischemia of the spinal cord.5

Referring-consultant feedback The plan agreed by the referring consultant was to have another trial with unipolar denervation with 18 G needle. The bipolar technique was agreed to be a good idea, but could not be attempted due to the non-availability of an appropriate RF machine. If the above fails, the plan was to try the cervical epidural injection of LA with nonparticulate steroid (Dexamethasone), using the catheter technique. Unfortunately, the patient was lost to follow-up; hence, a specific outcome could not be established.

References 1. 2.

3.

4. 5.

Lord SM, et al. Percutaneous radio-frequency neurotomy for chronic cervical zygapophyseal-joint pain. N Engl J Med 1996;335:1721–6. Sellon JL, et al. Cooled radiofrequency neurotomy for sacroiliac joint pain: A retrospective case review. PM and R. Conference: 71st Annual Assembly of the American Academy of Physical Medicine and Rehabilitation Seattle, WA United States. Conference Publication September 2010;2(9 Suppl. 1):S117–18. Cheng J, Pope JE, Dalton JE, Cheng O, Bensitel, A. Comparative outcomes of cooled versus traditional radiofrequency ablation of the lateral branches for sacroiliac joint pain. The Clinical Journal of Pain 2013;29(2):1536–5409. Langevin P, et al. Botulinum toxin for subacute/chronic neck pain. Cochrane Database Syst Rev. 2011;(7). Baker R, Dreyfuss P, Mercer S, Bogduk N. Cervical transforaminal injection of corticosteroids into a radicular artery: a possible mechanism for spinal cord injury. Pain 2003;103:211–15.

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this paper assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


Cervical meningioma in a patient with lumbar pain: a case report 1

Alan Fayaz and Serge Nikolic

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Abstract We present a case of a cervical spinal meningioma discovered in a patient with a seven-year history of lower-back pain with no abnormal neurological signs or symptoms. The case potentially calls into question the role and timing of imaging in both community and speciality assessment of lower-back pain, and highlights the importance of detailed history-taking. Keywords Cervical meningioma, lumbar pain, cervical spinal meningioma

Case history A 25-year-old female patient was referred to our chronic pain clinic by her GP in November 2011 with a seven-year history of back pain. The pain, described as a dull ache in her lower back, was episodic in nature with periods of up to five consecutive days’ absence of symptoms. The pain was worse in the morning and improved with movement. There was no significant past medical history with the exception of intraventricular haemorrhages at birth that had resulted in mild right-sided weakness, not apparent at the time of presentation. Her main concern was rationalisation of pharmacotherapy in the context of attempts to conceive. She was on tramadol 100 mg QDS, diclofenac 50 mg TDS, amitryptiline 50 mg OD and paracetamol 1 g QDS. She felt her pain had been best controlled when she was previously on a fentanyl patch at 12.5 mcg/hr. In the past she had experienced mild relief from a ten-session course of osteopathy, and six months of physiotherapy. She had tried TENS and Pilates with no tangible improvement. Examination was unremarkable with no facetal/intervertebral/SIJ tenderness and no abnormal neurology noted. The patient was reassured and sent home with minor alterations in her medication, to include re-initiation of her fentanyl patch, and a routine follow-up appointment. At six months her pain was noted to have improved on the patch, but was antagonised by prolonged muscle spasms. She also complained of intolerance to amitryptiline. A referral was made to our acupuncture service to help with the painful spasms and her amitryptiline was changed to duloxetine in the hope of improving compliance. Our patient was next seen in November 2012 (12 months after initial presentation) with modest improvement in her symptoms. It was felt that she would be an appropriate candidate for a pain management programme. Further questioning revealed that her pain had occasionally migrated upwards from her lower back to the infrascapular region. It also became apparent that she had never had any formal 1 2

Specialist Registrar in Anaesthesia and Pain Medicine, Barts Health NHS Trusts Consultant in Anaesthesia and Pain Medicine, Barts Health NHS Trusts Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


imaging of her spine and an MRI whole spine was requested. The MRI scan was performed a month later and the images showed a 16 x 14 mm intradural extramedullary mass in the cervical canal at level C3. There were signs of cord compression with local signal change and stretching of the adjacent roots. Her thoraco-lumbar spine was entirely normal. She was rapidly referred to our neurosurgical colleagues who performed an elective surgical resection. Histology confirmed a Schwannoma and postoperative imaging has confirmed complete removal of tumour tissue. She remains under the care of the chronic pain service; her lumbar pain has remained unchanged despite her surgery.

Discussion It has been estimated that 8 out of 10 people will be affected by back pain in their lifetime, with approximately 6% of these patients developing chronic or disabling pain.1 Primary spinal neoplasms have an incidence of 3–5 per million people per year and consequently represent a minor fraction of patients with back pain.2 Meningiomas account for up to 46% of primary spinal neoplasms, with less than 40% being restricted to the cervical spine; early identification and surgical resection is associated with favourable outcomes. It is difficult to ascertain whether the lumbar component of our patient’s back pain was related to her cervical tumour. The following were the reasons for ordering a spinal MRI scan: § § §

Persistent pain of eight years’ duration without any previous imaging of the spine. Occasional migration of our patient’s lower-back pain to the infrascapular region, which may imply cervical and/or thoracic pathology. Consideration for pain management programme.

A NICE guideline issued in 20093 restricts the use of MRI scan in non-specific lowerback pain (albeit of six weeks to 12 months’ duration, but this is often extrapolated to include all lower-back pain sufferers) unless a patient is considered for spinal surgery or malignancy, cauda equina, infection, fracture or inflammatory condition is suspected. We feel that the following points regarding spinal imaging in non-specific lower-back pain sufferers should be considered: § § §

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Spinal malignancy or indeed any other red flags may not have a typical presentation. Prolonged persistent pain where the adult is young should be an indication for imaging in its own right. Imaging should be considered for reassurance purposes, particularly in the context of proposing combined exercise therapy and psychological treatment programmes. The success of these programmes at least partly relies on patients’ acceptance of the fact that there isn’t any obvious ongoing damage – understandably, this is difficult to accept without evidence (such as an MRI scan).

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References 1

Jackson, M. A. & Simpson, K. H. Chronic Back Pain. Continuing Education in Anaesthesia, Critical Care & Pain 2006, 6(4):152–155.

2

Gottfried, O. N. Gluf, W., Quinones-Hinojosa, A., Kan, P., & Schmidt, M. H. Spinal meningiomas: surgical management and outcome. Neurosurgical Focus 2003, 14(6):e2, ISSN 1092-0684.

3

National Institute for Health and Clinical Excellence. Low back pain: early management of persistent non-specific low back pain (Clinical guideline 88) 2009, www.nice.org.uk/CG88 (accessed February 2013).

Contacts/correspondence Dr Alan Fayaz: alanfayaz@gmail.com

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

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Numbers needed to heal, numbers needed to harm, numbers needed to kill; reflections on opioid therapy and primary duty of medicine Dr Rajesh Munglani MB BS DA DCH FRCA FFPMRCA Consultant in Pain Medicine, Cambridge

I sometimes think that, having been called to practice in the Healing Arts, and in particular the Noble Art of Medicine, I feel like part of a wandering tribe in the desert. Nothing ever seems to stay the same. Whilst the human body and mind is probably more or less now what it was a few thousand years ago (perhaps a bit fatter from too much cereal consumption and soda pop and resulting in little more type 2 diabetes and cardiovascular disease – but that is another story), everything else to do with caring for the human condition changes at a frightening pace. Models of disease process and inferred practice that were considered acceptable and routine a generation or two ago are now considered to be grossly misleading and dangerous. I could mention bloodletting to quench fevers but a more telling story is the account of how many years it took for the medical profession to accept that the main cause of upper gastrointestinal ulceration was not too much acid but rather a bacterial infection. Between 1984 and 1989 two Australian scientists - doctors Warren and Marshall published five papers in highly reputable journals which conclusively showed that it was in fact a bacterium Helicobacter pylori rather than stomach acid that was the cause of gastrointestinal ulceration. Despite this the medical profession continued for many years to prescribe ineffective diets, antacid drugs and drastic surgery.1 At one point in 1988, frustrated at the deaf ears and unmoved hearts and minds, Dr Marshall infected himself with the Helicobacter pylori bacterium and of course promptly developed gastritis. It took approximately 5-10 more years before the medical profession finally came round to believing that the best way to treat stomach ulcers was to provide an antibiotic cocktail. Finally in 2005 Warren and Marshall were awarded the Nobel Prize for Medicine.

Why did it take so long for people to change in the face of new evidence? In one commentary on this subject, it states that people regarded as experts become used to doing something one way for decades and don't like to admit that another way, involving a paradigm shift, might be better. The commentators decided to call the systemic reticence to accepting the obvious change the “Warren and Marshall Syndrome”2. From my point of view it seems not quite the appropriate catchphrase,

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as Warren and Marshall were the ones who pushed the change rather than inhibited it. I am also reminded that in 1900 Lord Kelvin, then still –an undergraduate at Peterhouse Cambridge, famously announced that: “There is nothing new to be discovered …now. All that remains is more and more precise measurement.” 3 This was, of course, shortly before Einstein published his theories of relativity in 1905 and 19164. But the concern is that we, who regard ourselves as experts, naturally think we know most of what it is important to know about a subject. We therefore examine every new idea with a critical mind and, hopefully, with understanding, to realise if something truly new has been discovered. We need to be able to distinguish the useless snake oil from the highly effective tincture of opium, which may relieve the suffering of all mankind. However it does mean, on the flip side, that we as experts are less likely to accept novel but possibly true ideas if they don't fit the paradigms we have grown up with. We were taught that we are being told the absolute and incontrovertible truth when we were trained, such as fever can be eased by bloodletting, or that stomach acid causes ulceration. It has come as quite a shock to many of us in pain medicine that a drug that we had all considered to be relatively safe, has now been shown to be a major cause of morbidity and mortality both for the individual and society in general. Around 2004 we were just recovering from the Cox 2 inhibitor story5 6, thinking that initially this new type of inflammatory drugs was going to mean that we could provide anti-inflammatory medication and analgesia to our patients without the nasty side effects of stomach erosion, kidney failure etc. But then we found that they dramatically increase the chances of heart attacks and strokes, to the point where guidance from the medical authorities is now to virtually avoid using any Cox 2 inhibitor and indeed any NSAID except in the smallest dose for the shortest period, as they all share the same properties. Many of us switched to prescribing the seemingly safer opioids in view of the increasing dangers shown to be associated with anti-inflammatory medication. However there was another story that was unfolding, namely that there had also been a fairly aggressive increase in the use of opioids, particularly in the United States, with a ten- fold rise in opioid consumption in the twenty years since 1992 and in particular of the drug OxyContin, which was aggressively marketed as the extended version of oxycodone on release in 1995. Between 1997 and 2002 the use of oxycodone quadrupled7. But even at this time, clouds were looming over the "safe opioid story". Data was already emerging suggesting that the prescription of opioids was anything but benign. The National Centre for Health Statistics published a paper in 2009 reporting an increase in fatal poisonings in the United States between 1999 and 20068.

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Worryingly, the increasing death rate was particularly amongst younger people, as shown on the graph above. Overall the death rate tripled, particularly amongst young white males. Tis particular study specifically highlighted methadone but also other opioids as a particular drug associated with these deaths.

It was known that, for example, states like Florida had very high levels of death rate associated with the inappropriate prescribing - three times the rate of prescription of opioids compared to Illinois. One commentator stated that there was no evidence that people in Florida suffered more chronic pain than those in the rest of the United States. Another comment was that enough prescription painkillers were prescribed in 2010 to medicate every American adult round-the-clock for a month. In fact in the same year 2 million people reported using prescription painkillers non-medically for the first time - nearly 5,500 a day. Although most of these pills were prescribed for a medical purpose, many unfortunately ended up in the hands of people who misused or abused them. It was noted that most prescription painkillers were prescribed by primary care and internal medicine doctors and dentists, not specialists, and that roughly 20% of prescribers prescribe 80% of all prescription painkillers9.

The report also noted that:

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“… almost all prescription drugs involved in overdoses come from prescriptions originally. However, once they are prescribed and dispensed, prescription drugs were frequently diverted to people using them without prescriptions. More than three out of four people who misuse prescription painkillers used drugs prescribed to someone else.” The Centre for Disease Control published a paper in July 201110 stating that during 2003–2009, death rates increased for all substances except cocaine and heroin. The death rate from prescription drugs increased 84.2%, from 7.3 to 13.4 per 100,000 population. The greatest increase was observed in the death rate from oxycodone (264.6%), followed by alprazolam (233.8%) and methadone (79.2%). By 2009, the number of deaths involving prescription drugs was four times the number involving illicit drugs.

A further example came from West Virginia when it was noted that there was a 550% increase in mortality due to unintentional opioid-related overdoses between 1999 and 2004. Whilst it seemed initially that the majority of the problem was due to the diversion of opioid prescriptions to mainly young white males–2/3rds in fact - and this was often accompanied by ‘doctor shopping’11 until a prescription was obtained, it also became clear, however, that about 1/3rd of the mortality was actually in those patients who were prescribed the opioid in the first place.

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Thus this phenomenon of a massive rise in the marketing, sales and prescription of opioids was not just a problem of diversion, which could be seen as a simple (mainly US) sociological problem, but was also the cause of unintended mortality amongst patients who were being prescribed opioids. A study in Canada, where opioids are prescribed on state funding, without the financial incentives present in the US, showed that between 1997 and 2006, patients who were prescribed more than 200 mg of morphine a day were three times more likely to die as a consequence of the prescription12. In a study funded by the Veterans Administration13, Bonerht found the overall death rate to be in the order of 0.04% amongst those given an opioid prescription. The risk was in fact substantially greater with higher doses of morphine (above 100 mg), and was almost 10 times higher than if one was prescribed up to 20 mg per day. The risk was also substantially increased in those with a history of substance abuse and those on combined regular and p.r.n. prescription. Even accepting the lower overall risk of 0.04%, this worked out at one death in 2500 patients. The sharp eyed amongst you will have noticed that the figures above do not give any indication over what time frame the risk was present. In fact the authors worked out that the approximate average death rate per thousand chronic patient-months was approximately one. That is, one patient would die every month for every thousand patients taking an opioid prescription. By any account this is an extraordinarily high death rate, even higher when one takes into account doses above 100 mg of morphine equivalent, substance abuse and the type of prescription. This made me think about whether it was worth it? One could argue that if one person died, but 999 every month “thanked God for their doctor and their prescription” for the relief of their suffering, then one could argue that providing a quality of life to 999 out of every 1,000 patients per month isn't really that bad, as long as everyone was aware of the risk. After all, lots of treatments in medicine are risky. The chances of dying on the road every year for each one of those is approximately 1 in 17,000; the chances of an over 75-year-old on an antiinflammatory drug developing gastrointestinal haemorrhage is 1% per year, according to Bandolier14. However, what is the evidence that a prescription of opioids is effective for our patients in the first place? One of the studies above suggested that 3% of the American population were taking regular opioid prescriptions. Presumably this is for severe pain…. or is it? Is a complaint of pain a justifiable enough reason to give an opiate prescription? Would we have been negligent if we did not treat the pain and suffering of a fellow human being? It has been argued by Margaret Somerville, Professor of Law and Medicine at McGill University, that the unreasonable failure to provide adequate pain relief constitutes negligence15. Should we take patients at face value? If we did, we might be in trouble. In the Grampian region of Scotland, one study put the incidence of self-reported pain at over 50%16. On the face of it, other studies also support the very high incidence of self-reported musculoskeletal pain in the community, with a study from Europe giving rates of up to 50% or so for various pains, particularly with rising age17. However a further analysis in the same paper shows that whilst the incidence of general complaints of pain may be up to 40-50% depending on age group studied, the prevalence of clinically significant pain is much lower at about between 3-15%. More importantly it is crucial to recognise that perceived pain may be a symptom of another underlying diagnosis, or certainly exacerbated by it, in particular 13

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depression18. Indeed some authorities believe many cases of fibromyalgia are generally a manifestation of depression19. Failure to recognise that many factors may contribute to the final common presentation of pain is a profound mistake. The commonest error is to prescribe strong opioids in the presence of severe depression, distress, and complaints of pain, often on an unrecognised background of past history of significant psychological problems including abuse,20 drug and alcohol problems, in the vain hope that they will help generally. As one author put it “… Opioids are being used to treat this undifferentiated state of mental and physical pain.21” The presence of depression or anxiety has to be recognised in such patients and treated independently by pharmacological, psychological or other techniques22. The largest studies of opioid treatment of chronic pain suggest that, particularly in this distressed group of patients, improvements in either pain scores or quality of life are not achieved23. Becker et al (in Denmark, with the world’s highest rates of opioid prescription and consumption) suggest that those who went on to opioid therapy already had significant co-morbidity and were already consuming five times the health care resources prior to initiation of opioid therapy, and indeed already were more likely to have had pre-existing drug and alcohol problems24. Interestingly, however, Eriksen and his co-authors reported that addiction rates in their study patients (again in Denmark) were comparable to the general population25 Yet it is clear that subgroups of (correctly chosen) patients can do well with opioids on a number of measures, including addiction rates of less than 1% 2627282930and doctors with a little training can instinctively predict who are likely to do well31. On balance, carefully chosen opioid prescriptions in carefully chosen patients are unlikely to cause long term morbidity and mortality32. However, we need to recognise that currently the patients who end up on opioids may have severe pre-existing risk factors such as anxiety, depression, and addiction problems33, and quite simply, these patients need to be recognised and treatments planned more carefully for them34. These points have been highlighted by the Pain Society in their opioid prescribing and other guidelines35 and by Joan Hester in the BMJ36 It is also becoming increasing clear that not all opioids are the same. Diversion of opioids from their intended recipients is less likely to take place with sustained release patch formulation, particularly the newer opioid patch medications such as Butrans and Transtec (Buprenorphine patch) or Fentanyl patch (Durogesic patch), and problematic addiction problems and dose escalation is less likely with buprenorphine in particular. This, incidentally, is used to treat opioid addiction in some countries373839. However it is clear that further research is required40. What are the alternatives to prescription of analgesics? Talking therapies? Pain management programmes? Certainly unlikely to harm our patients, but time intensive and expensive. One study looking at the efficacy of practice based CBT found significant gains only in one in 4–7 patients41. A recent view in the Lancet by Turk et al42 makes very sobering reading as it states: “…that psychological treatment as a whole results in modest benefits in improvement of pain and physical and emotional functioning. ….evidence

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for the long-term effects is inadequate, and evidence is somewhat contradictory for effects on vocationally relevant outcomes” In fact the article isn't particularly positive about any pain treatment including any pharmacological or interventional therapies, stating that very roughly: “The best evidence for pain reduction averages roughly 30% in about half of treated patients, and these pain reductions do not always occur with concurrent improvement in function.” Against this modest benefit of active pain therapy the authors suggest that placebobased responses range between 10–20 per cent. The authors go on to state that this is hardly surprising in view of the complexity of chronic pain. In the absence of a cure we need to maximise pain relief so that patients are able to lead the highest quality of life possible. They also mention managing patient expectation but even more importantly they go on to state that because of the complexity of pain the tailoring of a treatment to an individual is paramount, and this is where future research should lie. We all have patients who have responded dramatically to one particular type of treatment or another. The real issue is how to avoid harming anybody else in the process of selecting the one who benefits. In the absence of a large group effect for any type of pain treatment, individual selection of patients is paramount. “Primum non nocere” First do no harm, and secondly show a degree of humility, not only as to the cause of what is troubling our patients, but how much we think we know about the treatment we are providing and how much good it will do. It is not that we get things wrong that is the problem, particularly when everybody else has made the same mistake, but it is a failure not to hear, when evidence is there in front of us, that we may have got things wrong, and to continue to insist on keeping deaf ears, hard hearts and closed minds. If the wandering tribe of Israel had been a little more open perhaps they wouldn't have had to wander around the desert for 40 years?

Conflict of interest disclosures

Conflict of interest: part of this article was submitted electronically as a letter to the BMJ. All mistakes, opinions expressed and misleading statements are entirely the responsibility of the author.

References 1

http://en.wikipedia.org/wiki/Helicobacter_pylori

2

Phinney and Volek The art and science of low carbohydrate living page 177

3

http://en.wikipedia.org/wiki/William_Thomson,_1st_Baron_Kelvin

4

http://en.wikipedia.org/wiki/Theory_of_relativity

5

http://en.wikipedia.org/wiki/Bextra

6

http://en.wikipedia.org/wiki/Vioxx

7

Okie, S.A flood of opioids, a rising tide of deaths N Engl J Med. 2010; 363:1981 -- 5

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8

Warner M, Chen LH, Makuc DM. Increase in fatal poisonings involving opioid analgesics in the United States, 1999–2006. NCHS data brief, no 22. Hyattsville, MD: National Center for Health Statistics. 2009

9

http://www.cdc.gov/homeandrecreationalsafety/rxbrief/

10

Drug Overdose Deaths — Florida, 2003–2009Weekly / Vol. 60 / No. 26 July 8, 2011The Morbidity and Mortality Weekly Report (MMWR) Series is prepared by the Centres for Disease Control and Prevention (CDC) and is available free of charge in electronic format. http://www.cdc.gov/mmwr/mmwrsubscribe.html.

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Hall et al. Patterns of abuse among unintentional pharmaceutical overdose fatalities JAMA. 2008; 300(22): 2613 -2620

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Gomes l et al. The opioid dose and drug related mortality in patients with non-malignant pain. Arch Intern Med. 2011;171(7):686 -- 691

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Bohnert et al Association between opioid prescribing patterns and opioid overdose deaths JAMA. 2011;305(13):1315 -- 1321

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http://www.medicine.ox.ac.uk/bandolier/booth/painpag/nsae/nsae.html#Heading10

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Brennan, Carr and Cousins. Pain Management: A Fundamental Human Right. Anesth Analg 2007;105:205–21

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Elliot, Smith, Penny, Smith and Chambers. The epidemiology of chronic pain in the community. Lancet 1999; 354:1248 -- 1252.

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Picavet and Schouten. Musculoskeletal pain in the Netherlands: prevalences, consequences and risk groups, the DMC3-study.Pain2003;102:167–178 18 Klauenberg, Maier, Assion et al. Depression and changed pain perception: Hints for a central disinhibition mechanism. Pain.2008;140: 332–343

19 Ahles, Yunus and Masi. Is chronic pain a variant of depressive disease? The case of primary fibromyalgia

syndrome. Pain.1987; 29 :105 -- 111 20

Balousek, Plane and Fleming. Prevalence of interpersonal abuse in primary care patients prescribed opioids for chronic pain. Society of General Internal Medicine 2007;22:1268 -- 73

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Sullivan, Edlund, Steffick and Unutzer. Regular use of prescribed opioids: Association with common psychiatric disorders. Pain.2005;119: 95 -- 103

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Gärtner · M. Schiltenwolf. Limited efficacy of opioids in chronic musculoskeletal pain. Analysis of cause. Schmerz 2004;18:506 -– 514

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Ballantyne. Opioids for chronic pain::Taking stock. Pain.2006:125:3 -- 4

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Becker, Thomsen, Olsen, Sjøgren, Bech and Eriksen. Pain epidemiology and health related quality of life in chronic non-malignant pain patients referred to a Danish multidisciplinary pain center. Pain.1997; 73:393.--400

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Eriksen, Sjøgren, Bruera, Ekholm and Rasmussen. Critical issues on opioids in chronic noncancer pain: An epidemiological study. Pain 2006;125: 172 __ 179

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Tassain V, Attal N, Fletcher D, Brasseur L. Long-term effects of oral sustained release morphine on neuropsychological performance in patients with chronic non-cancer pain. Pain.2003;104:389 -- 400

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Ballantyne and Fleisher. Ethical issues in opioid prescribing for chronic pain. Pain_2010;148:365 -- 367

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Noble, Tregear, Treadwell and Schoelles. Long-term opioid therapy for chronic non-cancer pain: A systematic review and meta-analysis of efficacy and safety. J Pain Symptom Manage 2008;35:214 -- 228

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Maier, Schaub, Willeber-Strumpf and Zenz. Long-term efficiency of opioid medication in patients with chronic noncancer-associated pain. Results of a survey 5 years after onset of medical treatment. Schmerz 2005;19:410 – 417

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Chou et al on behalf of The American Pain Society – American Academy of Pain Medicine Opioids Guidelines Panel Clinical guidelines for the use of chronic opioid therapy in chronic non-cancer pain. The Journal of Pain 2009:10:113 -- 130

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Miles J. Belgrade,* Cassandra D. Schamber,† and Bruce R. Lindgren. The DIRE Score: Predicting outcomes of opioid prescribing for chronic pain. The Journal of Pain 2006: 7;671 -- 681

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Jensen, Thomsen, Højsted. 10-year follow-up of chronic non-malignant pain patients: Opioid use, health related quality of life and health care utilization. European Journal of Pain 2006; 10:423 – 433

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Fields. Should we be reluctant to prescribe opioids for chronic non-malignant pain? Pain 2007;129:233 – 234

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http://www.britishpainsociety.org/pub_professional.htm

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Hester BMJ 2010;340:c476

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Jensen Thomsen, Højsted. 10-year follow-up of chronic non-malignant pain patients: Opioid use, health related quality of life and health care utilization. European Journal of Pain 2006;10: 423 – 433

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http://www.drugabuse.gov/Bupupdate.html

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Chou et al on behalf of The American Pain Society – American Academy of Pain Medicine Opioids Guidelines Panel Research gaps on use of opioids for chronic non-cancer pain: Findings from a review of the evidence for an American Pain Society and American Academy of Pain Medicine Clinical Practice Guideline. The Journal of Pain 2009;10:147 -- 159

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Morley, Williams and Hussein. Estimating the clinical effectiveness of cognitive behavioural therapy in the clinic: Evaluation of a CBT informed pain management programme. Pain 2008; 137: 670 – 680

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Turk, Wilson and Cahana. Treatment of chronic non-cancer pain. Lancet 2011; 377: 2226 – 35

Contacts/correspondence For example: Dr Rajesh Munglani, 64 Cambridge Road, Impington, Cambridge CB24 9NU Give your email here:

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2013. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

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Current understanding of the causes of Complex Regional Pain Syndrome (CRPS), with its MedicoLegal Implications Dr Andreas Goebel (pain medicine), PhD, FRCA FFPMRCA

1

Abstract Complex Regional Pain Syndrome (CRPS) is a painful post-traumatic condition affecting limbs. About one in seven patients will not get better even at 1–2 years after the eliciting trauma, and for this group establishing the causation can be an important issue within a medico-legal context. Known CRPS risk factors such as female sex explain only a small fraction of the variability in developing the condition after trauma, whereas recent research has soundly rejected the idea of a predisposing CRPS personality. Although it is unknown what causes CRPS, there is no scientific evidence to support the idea that any trauma at any time would have anyway caused the condition. In the absence of firm evidence on causation it appears most logical to consider the particular trauma sustained at the particular time point as the most crucial element to the CRPS causation. Keywords CRPS, Complex Regional Pain Syndrome, medico-legal, pain

Background CRPS typically develops after – often small – trauma to a peripheral limb.1 About 9% of patients cannot recall any trauma, or report CRPS-onset following everyday activities such as jogging or typewriting.2 Many people develop transient features of CRPS lasting for a few weeks particularly after fractures, which usually completely resolve.3,4 How is it possible that some patients develop a severe, lasting posttraumatic pain, given that these same patients, and indeed most of us experience small traumata many times in their lives without troublesome sequels?

The case The only correct answer to this, from my perspective, is that there is probably a reason, but we don’t know it. A popular default answer has been, for many years, that these patients have a psychological problem predisposing them to develop CRPS,5 but carefully conducted studies over the last five years have soundly rejected this option for the vast majority.6–8 Because of this strong refuting evidence, when patients do present with significant psychiatric, or psychological comorbidity, it should generally be assumed that this is either a chance coincidence (people with diabetes, for example, may also have psychiatric abnormalities, without suggestion of a link between their two problems), or that these psychological problems have 1

Pain Research Institute, Department of Translational Medicine, University of Liverpool; and the Walton Centre NHS Foundation Trust, Liverpool.

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developed in response to having CRPS; perhaps some patients are prone to react in that way to a major distress. So if there is no ‘CRPS personality’, what else could there be causing this perplexing and sad condition? Clues come from a recent population-based study, the first of its kind, which has demonstrated associations between CRPS and female sex, asthma, migraines, osteoporosis and ACE inhibitor intake.6,9 Thus, it is possible that people, particularly females with either a hereditary or a medication-induced tendency to develop augmented neurogenic inflammation2 after trauma, might be at risk. Nevertheless, the facts that most patients will have experienced uncomplicated traumata before their CRPS-triggering event, and, independently, that only very few patients will develop a second episode of CRPS (2–3% of all cases2) suggest that these epidemiological results do not sufficiently solve the puzzle of the CRPS causation; i.e. the development of CRPS is still unpredictable, but there are known risk factors explaining a small part of the observed variability after trauma. What are the implications of this current state of our knowledge in the medico-legal context? It appears that we indeed have to attribute causation to the specific trauma sustained. The alternative idea, that a patient would have developed CRPS ‘anyway’ with any subsequent trauma, would not appear logical given the usually unremarkable medical history as discussed above. It appears to be a logical approach if we assume that a majority of patients go through a ‘time-window of vulnerability’ created by unknown, mostly biological factors, and if a trauma occurs during this time, the risk for CRPS development is high: neither earlier, nor later trauma would then be equally detrimental. Thus, the trauma, occurring at the particular time at which it occurs should be important. A novel twist to this story is provided by both ours and some other groups’ recent research results.10–12 These results suggest that CRPS may in fact be a post-traumatic, regional, non-destructive auto-immune disorder, a prototype perhaps of a novel kind of auto-immunity.13 We hypothesise that an underlying auto-antibody-mediated auto-immune reaction, which is transient in the majority of patients, creates transient vulnerability to trauma. Transient auto-antibody-mediated disease is not uncommon in medicine (an example is Guillain-Barre syndrome); however, autoantibodies here are usually understood to be both essential and sufficient to cause disease, while we suggest that, in CRPS, trauma is required in addition to a raised serum-auto-antibody level (i.e. auto-antibodies are essential, but not sufficient). Most CRPS is transient (patients get better, although – with the exemption of those very early cases mentioned above – many will suffer important sequels), but in the medico-legal context chronic CRPS (patients whose pain does not get better at all) is probably more of a concern. We know now, from the same epidemiological study discussed above, that most patients will improve (although not necessarily fully 2

‘Neurogenic inflammation’ is an inflammatory response sustained by substances antidromically (i.e. against the normal course) released by peripheral sensory nerves. These substances include ‘Substance P’ (SP), and ‘Calcitonin Gene Related Peptide’ (CGRP) and others. The fact that CRPS is associated with ACE inhibitor intake (ACE inhibitors block neuropeptide metabolism), but not with the intake of any other antihypertensive medications further supports the notion of an important role of neuropeptides in CRPS. Neuropeptides cause swelling, reddening, and warmth, but other CRPS signs, such as sweating, can probable not be explained by their action and should have other causes.

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recover) within the first 6–24 months after disease onset.14. Only one in seven patients will not improve by 24 months, and this group is less likely to quickly get better thereafter. How are these patients with chronic disease biologically different from those who got better? In order to answer this question, considerable research effort has focused on the idea that, as pain becomes chronic it may be mostly sustained by the brain, rather than by any peripheral (limb) processes. Thus, it is proposed, that in susceptible patients, after an initial painful peripheral traumatic insult, in the long run neural cell networks in the temporal brain lobe and elsewhere will retain an abnormal pattern of activity, sustaining a perception of pain; even so the peripheral insult has completely resolved. These ideas have gained further credence from the fact that in most patients with chronic CRPS limb disease signs, i.e. what we can see – swelling, colour changes, hair and nail growth changes, sweating – will reduce with time, while the only remaining abnormalities are often the patients’ reported un-abiding pain and inability to fully move the affected limb.15 Thus, again, the idea here is that any original (if poorly understood) post-traumatic augmented peripheral inflammatory process reduces with time, and the central nervous system takes over. A Central Nervous System-sustained pain would resemble what has been shown in phantom limb pain3.16 Of note, this concept has not been proven in CRPS. Although patients with CRPS show substantial and important abnormalities, termed ‘cortical reorganisation’, in their brain’s processing of peripheral signals, we don’t know whether these processes are in fact hen, or egg, i.e. whether they really can sustain CRPS on their own, or whether they are themselves dependent on unknown on-going peripheral processes.17 Contrasting the popular idea of the importance of a central driver for long-standing CRPS, we suggest that in this group auto-antibodies are still essential pathogenetic elements: auto-antibody serum-titres will not recede, but these proteins will remain in the systemic circulation, similar as is the case in some chronic, auto-antibodymediated auto-immune diseases, such as CIDP,18 but unlike in the acute autoantibody-mediated auto-immune diseases such as Guillain-Barre syndrome where these auto-antibodies eventually disappear. These patients with long-standing CRPS may have an on-going, active auto-antibody-mediated immune process sustaining the clinical condition. In consequence, this process may in fact be treatable with immune treatments, even in very long-standing cases. Thus far, this is a largely unproven proposition. We have shown that some patients with long-standing CRPS respond dramatically with pain reduction to ‘polyvalent immunoglobulin’, an immune treatment, potentially supporting the idea.19

3

Phantom limb pain is pain in a non-existing arm, or leg, for example after traumatic amputation.

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Figure 1

Simplified hypothetical relationship between autoantibody titre, CRPS disease signs, and pain intensity. A quickly resolving CRPS: if limb trauma occurs at a time (window of vulnerability, wv) where patients have a high titre of relevant serum auto-antibodies (green dashed line), then a post-traumatic reaction ensues, with disease signs such as enhanced swelling, and temperature changes, blue line dash-dot, and high pain intensity (red line), fulfilling diagnostic criteria for CRPS; however, as the autoantibody (AAB) titre naturally normalises, so do these CRPS features; B un-resolving CRPS: here, the auto-antibody titre only partially normalises, and while the initial posttraumatic reaction may slowly reduce through natural anti-inflammatory mechanisms, binding auto-antibodies continue to sustain the condition. Orange dotted line: normal course of swelling and pain after radius fracture. Adapted from reference 13, with permission.

Thus, auto-antibodies, after their first appearance in the body (presumed – as in other auto-immune diseases – to often occur after infections which abnormally activate the immune system) will then continue to be produced for a long time, or even indefinitely, and will sustain the CRPS clinical presentation. Although one might consider that once a person has acquired such serum-auto-antibodies, any trauma could elicit CRPS, the fact is, that even people with long-standing CRPS will only very rarely develop CRPS in a second limb after additional trauma. We propose that the level of auto-antibodies required to sustain CRPS is lower than that required to elicit CRPS after trauma (Figure 1 B). It appears we won’t get away from the presumption that the incident trauma sustained at a particular time is probably essential for the CRPS disease process, and that even in these patients who develop long-standing CRPS, a trauma sustained three weeks later, or earlier, may not have had the same effect. What, then, about those rare patients with chronic CRPS, who develop pain in additional limbs, without additional trauma (about 7% of cases20) – can these additional problems really be attributed to the original trauma? First, in these cases it remains to be seen whether the now newly-affected limb is really affected by ‘spreading’ CRPS.4 The new Budapest criteria for CRPS have now been accepted by the International Association for the Study of Pain (IASP),21 and in consequence in order to confirm the CRPS diagnosis patients should have disease signs such as limb swelling or sweating; i.e. it is now not sufficient for making a diagnosis of spreading 4

‘CRPS spread’ is the development of CRPS in body parts not initially affected; for example, where initially only one hand is involved, over time the whole arm and shoulder become affected (‘proximal spread’), or other limbs become affected, either following additional trauma, or spontaneously.

21

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when the patient reports such abnormalities to occur occasionally at home, while the doctor can’t see them in clinic. In my experience, such signs are often not present in presumed ‘spreading CRPS’. The Budapest criteria allow the use of the term ‘CRPS NOS’ (‘not otherwise specified’) in these cases. However, let’s be clear that we are now talking about a very small subgroup within an already rare condition, and unfortunately little evidence exists to guide us. We can perhaps be forgiven, in the medico-legal situation, to be uncertain about the causation of any spreading in these cases.

Summary As recent research evidence has helped us to nudge closer to gaining an understanding on the causes of CRPS, this has not changed the fact that the incident trauma at the specific time when it occurred should remain the single most important discernable causative factor for most patients; there is little support for a role of any CRPS-preceding psychopathology, or for the notion that just about any trauma sustained at any time would have caused the same condition. Watch this space …

Conflict-of-interest disclosures

The author provides medico-legal reports for cases of post-traumatic CRPS for both plaintiff and defendant. He has received research support from CSL-Behring, BPL, Biotest, Talecris, Baxter and Grifols, Pfizer; travel support from CSL-Behring and Baxter; speaker honoraria from Baxter; and consultancy fee from Biotest.

References 1. Goebel A. Complex regional pain syndrome in adults. Rheumatology (Oxford) 2011;50:1739. 2. Veldman PH, Reynen HM, Arntz IE, Goris RJ. Signs and symptoms of reflex sympathetic dystrophy: prospective study of 829 patients. Lancet 1993;342:1012. 3. Atkins RM, Duckworth T, Kanis JA. Algodystrophy following Colles' fracture. J Hand Surg. Br. 1989;14:161. 4. Dijkstra PU, Groothoff JW, ten Duis HJ, Geertzen JH. Incidence of complex regional pain syndrome type I after fractures of the distal radius. Eur J Pain 2003;7:457. 5. Verdugo RJ, Ochoa JL. Abnormal movements in complex regional pain syndrome: assessment of their nature. Muscle Nerve 2000;23:198. 6. de Mos M, et al. Medical history and the onset of complex regional pain syndrome (CRPS). Pain 2008;139:458. 7. Beerthuizen A, et al. The association between psychological factors and the development of complex regional pain syndrome type 1 (CRPS1) – a prospective multicenter study. Eur. J Pain 2011;15:971. 8. Beerthuizen A, van 't SA, Huygen FJ, Klein J, de Wit R. Is there an association between psychological factors and the Complex Regional Pain Syndrome type 1 (CRPS1) in adults? A systematic review. Pain 2009;145:52-59. 9. de Mos, MM, Huygen FJ, Stricker BH, Dieleman JP, Sturkenboom MC. The association between ACE inhibitors and the complex regional pain syndrome: Suggestions for a neuro-inflammatory pathogenesis of CRPS. Pain 2009;142:218-24. 10. Kohr D, et al. Autoimmunity against the beta(2) adrenergic receptor and muscarinic-2 receptor in complex regional pain syndrome. Pain 2011;152: 2690-700. 11. Goebel A, et al. Immune responses to Campylobacter and serum autoantibodies in patients with complex regional pain syndrome. J. Neuroimmunol. 2005;162:184-9. 12. Blaes F, et al. Autoimmune etiology of complex regional pain syndrome (M. Sudeck). Neurology 2004;63:1734. 13. Goebel A, Blaes F. Complex Regional Pain Syndrome, prototype of a novel kind of autoimmune disease. Autoimmunity Reviews 2013; 12(6):2690-700. 14. de Mos, MM, et al. Outcome of the complex regional pain syndrome. Clin. J Pain 2009;25:590-7. 15. Birklein F, Riedl B, Sieweke N, Weber M, Neundorfer B. Neurological findings in complex regional pain syndromes – analysis of 145 cases. Acta Neurol. Scand. 2000;101:262. 16. Flor H, Nikolajsen L, Staehelin JT. Phantom limb pain: a case of maladaptive CNS plasticity? Nat. Rev. Neurosci. 2006;7:873. 17. Maihofner C, Handwerker HO, Neundorfer B, Birklein F. Cortical reorganization during recovery from complex regional pain syndrome. Neurology 2004;63:693. 18. Gorson KC, Allam G, Ropper AH. Chronic inflammatory demyelinating polyneuropathy: clinical features and response to treatment in 67 consecutive patients with and without a monoclonal gammopathy. Neurology 1997;48:321. 19. Goebel A, et al. Intravenous immunoglobulin treatment of complex regional pain syndrome: a randomized trial. Ann. Intern. Med. 2010;152:152.

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20. Maleki J, LeBel AA, Bennett GJ, Schwartzman RJ. Patterns of spread in complex regional pain syndrome, type I (reflex sympathetic dystrophy). Pain 2000;88:259. 21. Harden RN, et al. Validation of proposed diagnostic criteria (the 'Budapest Criteria') for Complex Regional Pain Syndrome. Pain 2010;150:268.

Correspondence Dr Andreas Goebel, Pain Research Institute, Department of Translational Medicine, University of Liverpool, Liverpool L9 7AL

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2013. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

Â

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Stent-Mediated Redistribution of Cerebral Venous Outflow in the Treatment of Severe Intractable Headache: A Case Report *

Nicholas Higgins FRCR, Frances Hall FRCP, John Pickard F Med Sci

Abstract We describe a patient with multiple symptoms but whose primary complaint was of headache, in whom no firm diagnosis was made in two years, who was resistant to all treatment, until a markedly asymmetrical cranial venous outflow came to be regarded, not as normal variant anatomy but as fundamental to the clinical problem. Deliberately altering this anatomy in favour of a more symmetrical arrangement by stenting a hypoplastic transverse sinus brought about an immediate, profound and sustained clinical improvement. This result challenges the existing consensus on what is acceptable as normal in respect of cranial venous outflow. It raises intriguing questions about the relationship between neurological symptoms and the vagaries of cranial venous outflow anatomy. It suggests there may be new opportunities in the investigation of chronic headache. Keywords Headache, epistaxis, cerebral venous outflow, venous sinus anatomy, venous sinus stenting

Introduction Short of venous sinus thrombosis, the suggestion that neurological symptoms or syndromes might be attributed solely to venous disease is generally met with scepticism or outright disbelief.1,2,3 Yet, if cerebral venous outflow obstruction from venous sinus thrombosis can produce a florid and sometimes fatal neurological illness, there is no reason, in theory, why lesser degrees of venous obstruction should not produce a more subtle neurological disorder. What is more, given the complexity of the clinical syndrome that may result from frank venous sinus thrombosis,4 the clinical correlate of a lesser degree of venous obstruction might not be easy to recognise. Add to this a wide acceptance of almost any appearance of the venous sinuses and neck veins as part of normal anatomical variation,5,6,7,8 then attributing symptoms to a particular venous anatomical arrangement becomes almost impossible. Against this background, headache clinics are overwhelmed with patients whose diagnoses are either unknown or comprise little more than a description of their symptom complex with no pathological substrate; this usually confirmed by normal blood tests and normal radiology.9,10 Could any of these patients have venous disease and how would you prove it if they did? We describe a patient with severe debilitating * † ‡

Consultant Neuroradiologist, Addenbrooke’s Hospital, Cambridge, UK. Consultant Rheumatologist, Addenbrooke’s Hospital, Cambridge, UK. Professor of Neurosurgery, Addenbrooke’s Hospital, Cambridge, UK. Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


headache and subjective neurological symptoms who was undiagnosed for two years until investigations were specifically targeted at the cerebral venous circulation. Treatment resulted in a profound clinical improvement, which has been sustained over more than two years of follow-up.

Case report Initial history and investigations A 33-year-old woman was referred with headache and multiple symptoms, having already been extensively investigated the by neurology services at several other institutions. She gave a history of being completely well until she had been woken from sleep 18 months earlier with severe bilateral retro-orbital headache, at first associated with nausea and vomiting. There had been no head injury, alcohol binge or any other obvious precipitant. This headache had been present constantly since its first appearance and had been unresponsive to ordinary analgesics, pizotifen, amytriptyline, topiramate and labetolol. Opiate patches provided some relief. Two months after the onset of headache she had begun to complain of visual disturbance; at first, a dark spot in the inferotemporal field of the left eye, eventually progressing to a substantial bitemporal hemianopia. At the same time, she had noticed variable numbness in her left face, arm and lower leg with occasional similar symptoms on the right. Eight months after the onset of headache she had begun to experience frequent and recurrent nose bleeds. Sometimes, these were preceded by a reddish discoloration over her left cheek and they were followed by a period of confusion, dizziness and difficulty speaking. Around the same time, she had noticed mild weakness in her left arm, which meant she could not hold up a cup or kettle, or use a pen on that side (she was normally ambidextrous). Twelve months into her illness she noticed that her periods had become irregular; her hair growth had slowed; and she was suffering from poor memory, fatigue, dry skin, dry eyes, dry mouth and dry nose. (Figure 1). Figure 1 – Timeline of symptom development

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Months from onset of illness

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On examination, she was slim and looked well. She was mildly hypertensive when first seen but normotensive subsequently. She exhibited a mild Raynaud’s phenomenon and a subtle livedo reticularis rash over her knees and ankles but there were no other significant non-neurological findings. Her optic fundi were normal. Visual fields to confrontation at first appeared to confirm a bitemporal hemianopia but there were inconsistencies in her response to testing that cast doubt on this finding. Other cranial nerves were normal. There was a mild, non-pyramidal weakness of the left arm with no accompanying upper motor neuron signs. There was impairment to light touch up to the left ankle and throughout the left arm to the shoulder. Brain CT had been normal. Brain MRI had been normal except for a 7 mm nodule in the right pituitary. This nodule had minimal impact on pituitary volume and was unchanged over a four-year period. There was no suprasellar extension and no compression of the optic chiasm. MR venography had been unremarkable, though showing marked dominance of the left transverse sinus (Figure 2). Blood haematology and biochemistry had been normal. Full endocrine testing had been unremarkable, except for intermittently positive antinuclear antibodies. CSF pressure was unremarkable and biochemistry had been normal. Visual evoked responses and multifocal electroretinography had been normal. Specialist ear, nose and throat evaluation, including nasal endoscopy, had not established a cause of her epistaxis. Figure 2 – Magnetic resonance venograms

SSS

SS

(a) Left-hand image gives a lateral view of a normal superior sagittal sinus (SSS) and straight sinus (SS). (b) Righthand image gives an axial view through the posterior fossa showing marked dominance of the left transverse sinus (large arrow). The right transverse sinus is patent but small (little arrows). (There is some contamination of both images by signal from the carotid and vertebrobasilar systems.)

No neurological diagnosis was reached. A rheumatology opinion was sought and the possibility of a connective tissue disorder discussed, with primary Sjogren’s the most likely contender. However, there was a consensus that a unifying diagnosis which would account for the whole clinical picture was unlikely to be found, leaving symptomatic treatment the only therapy on offer.

Investigation of cerebral venous outflow Her symptoms progressed, especially epistaxes, which were now occurring several times a week and often formed part of a stereotyped response to a particular posture. Bending forwards to wash her hair, for example, markedly exacerbated her headache. 26

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This was often followed by flushing and fullness in her face, which was usually followed, in turn, by brisk epistaxis lasting up to 20 minutes. At the same time, whether or not she developed epistaxis, she would often experience a period of confusion, dizziness and difficulty with speech lasting about 45 minutes. She was unable to work (Figure 3). Figure 3 – Timeline of a typical episode of epistaxis and confusion

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15 In

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45

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Time in minutes from symptom onset

About this time she reported an intermittent rushing noise on the left side of her head, more noticeable when she lay on her left side. There was no audible bruit but to exclude an underlying vascular cause for her symptoms she was referred for catheter angiography. The angiogram showed normal arterial anatomy. Venous anatomy was also within accepted normal limits, although there was very marked dominance of the left transverse sinus which took the bulk of venous outflow from the sagittal and straight sinuses (Figure 4). The left jugular vein was also very much larger than the right (Figure 5).

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Figure 4 – Right carotid angiogram (frontal view)

The arterial phase (left image). The venous phase (right image) shows venous outflow almost exclusively to the left side.

Figure 5 – Right carotid angiogram (frontal view, centred over skull base and neck)

RIJV

LIJV

The left internal jugular is normal (LIJV). The right internal jugular vein is small (RIJV). There is faint opacification of small right transverse and sigmoid sinuses (arrows).

Looking to test the significance or her particular venous anatomy, gentle manual compression of the right side of the neck produced no response. Gentle compression of the left side, however, caused her face to become suffused with the colour puce. This receded immediately compression was released but was followed by a period of mild confusion and disorientation, difficulty with speech and left facial numbness which lasted about 45 minutes. Once she had recovered she was able to attest to the similarity of this episode with those she had been experiencing at home. She was taken back into the angiography suite for catheter venography. This procedure, performed under local anaesthesia, involves examination of the venous sinuses and jugular veins using a catheter passed cranially from a common femoral 28

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puncture. Injection of radiographic contrast medium allows visualisation of the sinuses and the direction of flow in them. Attaching a transducer to the catheter hub allows the recording of intrasinus pressures (Figure 6). Figure 6 – Catheter venogram, frontal view

SSS RTS LTS

RIJV LIJV

Left image shows radiographic contrast injected through a catheter in the sagittal sinus (SSS) draining exclusively into the left transverse sinus (LTS) and then the left internal jugular vein (LIJV). Right image shows a contrast injection from a catheter positioned in the right transverse sinus (RTS): the right transverse and sigmoid sinuses are small, as is the right internal jugular vein (RIJV).

Under baseline conditions, sagittal sinus outflow was exclusively to the left side; the right transverse sinus being patent but narrow and with minimal antegrade flow (Figure 6). Sagittal sinus pressure was recorded at baseline (9 mm Hg), then with right jugular compression (11 mm Hg), then with left jugular compression (29 mm Hg) (figures 7 and 8). Right atrial pressure was 5 mm Hg. This meant that a 24 mm Hg pressure gradient developed between the sagittal sinus and right atrium with left jugular compression. Further measurements showed that most of this gradient (14 mm Hg) was along the transverse sinus. Her symptoms after jugular compression developed as before but she was able to cooperate with the procedure and was fully recovered in about 45 minutes.

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Figure 7 – Continuous pressure recording from the sagittal sinus during right jugular compression

30mm Hg 20 10 0 0

5

10

15

secs 20

Drawn arrow marks beginning of right jugular compression lasting 15 seconds. Sagittal sinus pressure increases by about 2 mm Hg during that period, then returns to baseline once compression is released.

Figure 8 – Continuous pressure recording from the sagittal sinus during left jugular compression

30mm Hg 20 10 0 0

5

10

15

secs 20

Drawn arrow marks the beginning of left jugular compression lasting about 15 seconds. Sagittal sinus pressure increases gradually by 20 mm Hg during that period, then returns to baseline once compression is released.

Treatment by venous sinus stenting By this time it was more than two years since the onset of symptoms and she had not worked for six months. Therefore, with no alternative diagnosis on offer, we elected to proceed on the basis that her clinical syndrome was a consequence of the observed asymmetry in cranial venous outflow. There was no evidence of previous sinus thrombosis and the right transverse sinus was presumed congenitally hypoplastic. This meant there was no indication of what might have precipitated the onset of symptoms. Nevertheless, the hypothesis for her current condition was that she was so highly dependent on left-side cranial venous outflow that whenever the left jugular vein was compressed, she developed symptoms of venous compromise and raised intracranial pressure, of which headache, confusion and disorientation were one expression and epistaxis another. She was booked for stenting of the right transverse sinus with the aim of facilitating an alternative pathway for cranial venous outflow whenever flow in the left jugular vein was compromised. The right transverse sinus was small but still provided a potential conduit for venous outflow from the sagittal sinus in these circumstances, 30

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although currently only at the cost of a substantial pressure gradient. Stenting would enlarge this conduit to allow a greater flow of blood with a smaller pressure gradient under the same conditions. Stenting was performed under general anaesthesia using a right jugular puncture. In the procedure a 5F guide catheter was passed into the right sigmoid sinus under Xray control and a guide wire passed across the right transverse sinus into the sagittal sinus. This was used to deploy 4 mm diameter stents (Pharos; Micrus Corp) along the whole of the right transverse sinus under heparin anticoagulation (Figure 9). Figure 9 – Frontal view venogram after stent placement in right transverse sinus

RTS

RIJV

RTS

LTS

RIJV

Left image: unsubtracted film showing the stents along the right transverse sinus (arrows). The stent delivery catheter can be seen entering the sigmoid sinus from the right internal jugular vein. Right image: subtracted view in same projection following injection of the sinus with radiographic contrast. There is antegrade flow from the (stented) right transverse sinus (RTS) into the right jugular vein (RIJV) and retrograde flow across the confluence of sinuses into the left transverse sinus (LTS).

She had some headache localised to the site of the stents the next day but, otherwise, she described her head as feeling better than it had done at any time since she had become ill. Under supervision she bent forwards, in the same way that would previously have produced an immediate exacerbation of symptoms, without incident. She was discharged with instructions to take aspirin 75 mg and clopidogrel 75 mg daily for three months as a precaution against thromboembolic complications. At six-week follow-up she said that her quality of life was greatly improved. Rightside headache (from the stents) had resolved. She still had some generalised headache but this was much less severe than before stenting. Bending forwards still made her headache worse, although not as dramatically as before. She had had no further epistaxes and no further episodes of confusion. Normal function had returned to her left arm. Repeat catheter venography showed baseline pressure in the sagittal sinus similar to that previously (11 mm Hg). This increased to 19 mmHg with left jugular compression; that is much less than before and now not accompanied by any change in her clinical status. Pressure measurements undertaken in the right lateral sinus during left jugular compression (diverting most cranial venous outflow through the 31

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right side) now showed only a 1 mm Hg gradient along the stented right transverse sinus but there remained a 4 mm Hg gradient along the right sigmoid sinus. She was admitted for a second stenting procedure, this time of the rest of the right lateral sinus, effectively extending the previous stents through the right sigmoid sinus into the jugular bulb. This produced a further improvement in headaches. She had all the stents dilated up with a balloon in a third procedure, after which her headache virtually resolved. Subsequent CT and catheter venography (13 months after the first stenting procedure) showed all the stents widely patent (Figure 10) and sagittal sinus pressure increasing by only 4 mm Hg in response to left jugular compression. Figure 10 – Follow-up CT venogram of the posterior fossa

RTS LTS

RSS

RSS

LSS

Axial scans at different levels showing contrast opacification inside the stents (arrows). Left-hand image shows stents in the right transverse sinus (RTS), middle image in the right sigmoid sinus (RSS) and right-hand image at right sigmoid sinus outflow into the jugular vein. Note the normal transverse and sigmoid sinuses sinuses on the left side (LTS, LSS).

In two years of follow-up there have been no further epistaxes and no further episodes of confusion. She still complains of restricted vision but said there had been some improvement since the start of treatment (her visual fields never having been recorded as abnormal on formal testing). Her memory had returned to normal. She still suffers with dry eyes, mouth and nose. Her periods are regular but short. She is still fatigued but this is less disabling than previously. She is back at work full time. She is off all medication.

Discussion With regard to chronic headache, clinicians instigate scans in the strong expectation that the results will be normal and radiologists view the images on the understanding that symptoms rarely have any basis in a structural lesion.9,11 The exceptions (tumour and hydrocephalus, for example) are usually not difficult to detect. This understanding between these two medical disciplines may seem entirely appropriate at first sight. However, a critical look at the way in which radiologists arrive at a diagnosis of normality when reviewing a brain scan suggests that a scan declared as normal in these circumstances, in reality, brings little independent evidence to the clinical diagnosis. Thus, it is probably self evident that radiologists develop much of their expertise by reviewing large numbers of images, both normal and abnormal. This allows the accumulation of a mental database of normal appearances for use as a reference 32

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when differentiating significant pathology from what is irrelevant and what is normal variant anatomy. Studies on normal volunteers are available12,13,14 but in practice this database is largely derived from patients whose scan findings, whatever they might be, are assumed to be unrelated to the illness under investigation. This might be reasonable in the case of a patient who is asymptomatic and has a brain scan looking for metastatic cancer, for example. Here, any appearance out of the ordinary which is not tumour could reasonably be ascribed as incidental or a variant of normal. However, it might not be reasonable to make the same deduction in the case of a patient complaining of a headache. In this instance, especially if there was no pathological diagnosis, how could you be sure that whatever you had seen on the scan was not contributing to symptoms? The question is pertinent because patients with headache contribute substantially to the workload of any radiology department and, therefore, substantially to this mental database of normal appearances. With respect to the venous sinuses, this gives rise to a problem encapsulated in attempts to determine normal, in vivo anatomy. Ayanzen et al.15 performed MR venography in 100 patients whose MRI brain scan had already been diagnosed as normal, looking to catalogue the gamut of normal variant appearances for reference purposes. They found flow gaps in 31% of transverse sinuses, most of them in nondominant sinuses, but a few in co-dominant sinuses, and argued that these should not be mistaken for sinus disease, a finding accepted largely without question by both clinicians and radiologists. Taken for granted in this work, however, is that flow gaps in the venous sinuses could not possibly be responsible for symptoms. Yet, if one considers the commonest symptom and most frequent diagnosis in patients with a normal MRI scan – headache of unknown cause16 – then this assumption becomes highly questionable. Moreover, if one takes the trouble to recruit a control group which is headache-free then the presence of flow gaps in the venous sinuses takes on an entirely new and important significance. Higgins et al.,17 for example, compared the appearances of the venous sinuses in patients with idiopathic intracranial hypertension with those of normal volunteers specifically screened for the absence of headache. They found flow gaps in both transverse sinuses in more than 50% of patients with idiopathic intracranial hypertension but in none of the controls. Using ‘supernormal’ controls in this way, therefore, gave birth to a radiological sign of raised intracranial pressure (bilateral transverse sinus flow gaps) hitherto completely submerged by the incorporation of patients, whose symptoms should have excluded them, into the collective understanding of normative appearances. On debatable grounds, therefore, radiologists are using patients with chronic headache, in whom the scan does not show any obvious structural abnormality, as their yardstick of what is acceptable as normal. They are encouraged in this view by clinicians who cite the frequency of a normal scan in such cases as evidence that most chronic headache does not have a basis in a structural lesion. What this means is that, without recognising it, radiologists and clinicians are colluding in a circular argument, effectively excluding common observations from being implicated in a common complaint. The same concerns apply to the concept that asymmetry is a normal condition of cranial venous outflow. This idea has been developed over many years of cerebral angiography independently of CT or MRI. Yet, here as well, the observations on normal anatomy are derived from patients who are ill and have to be judged in that context. Much of angiography, for example, is undertaken in the investigation of headache with or without proven intracranial haemorrhage. Often, no cause of 33

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symptoms or haemorrhage is found and often there is marked asymmetry of cranial venous outflow through the transverse sinuses. This almost never invites comment and is usually accepted as part of normal anatomical variation.18 Yet, how can one be sure that the appearance of the venous sinuses in these cases is irrelevant? It is widely recognised, for example, that some cases of subarachnoid haemorrhage have a venous aetiology,19 reason enough by itself, to disqualify any of these patients from forming the basis of our understanding of normal venous anatomy. More recent attempts to document the asymmetry or otherwise of venous sinus outflow have focused on MR venography, again with asymmetry, sometimes quite marked, a common finding.6,7,8 These studies all used the fact of a normal MRI as a licence to interpret any appearance of the venous sinuses as part of normal variant anatomy. Yet, MRI in all these studies was performed in the investigation of symptoms, not always specified, making their conclusions with respect to the venous anatomy equally suspect. In the case we describe, no progress was made in diagnosis or treatment until the observed asymmetry of cranial venous outflow came to be viewed as part of the problem rather than as simply an expression of normal venous anatomy. The catalyst for this change of view was a somewhat speculative clinical test in which each of the internal jugular veins was compressed in turn and the result observed. This is not a manoeuvre described in the medical literature, except with reference to tinnitus in association with idiopathic intracranial hypertension.20 Nevertheless, its outcome here established a connection between symptoms and the particular venous anatomy. Then, having accepted this connection, partial restoration of outflow symmetry by stenting a hypoplastic transverse sinus brought about an immediate and profound symptomatic improvement. There were no further epistaxes, for example, despite instigation of dual antiplatelet treatment to maintain stent patency. Repeats of the jugular compression test no longer produced any adverse clinical symptoms. Subsequent procedures, in which the outflow asymmetry was further reduced, gave further incremental reductions in headache. There has been no deterioration in two years of follow-up. Unexplained, of course, is what precipitated the clinical syndrome in the first place in a patient whose cerebral venous outflow anatomy appeared to be congenitally determined. Unexplained also is why headache was constant (though variable in intensity) when intracranial venous pressures and CSF pressure were normal in the resting state, although in this regard, the time course (45 minutes) of symptoms precipitated by a short period of left jugular vein compression is intriguing. These questions aside, this case raises the possibility that asymmetrical cerebral venous drainage, particularly highly asymmetrical venous drainage, may be very unusual in the normal population. It suggests that, in the context of headache and unexplained symptoms, it may be inappropriate to write off such appearances as simply a manifestation of normal variant anatomy without clinical significance. It invites speculation that there may be an explanation in the venous anatomy for symptoms in other headache syndromes; although, without the insight that was afforded in this particular case by the jugular compression test, the diagnosis is unlikely to be easy.

Conflict-of-interest disclosures

There are no conflicts of interest. This work was unfunded. 34

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References 1.

Baracchini C, Atzori M, Gallo P. CCSVI and MS: no meaning, no fact. Neurol Sci. 2012 May 9 (Epub ahead of print).

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Khan O, Tselis A. Chronic cerebrospinal venous insufficiency and multiple sclerosis: science or science fiction? J Neurol Neurosurg Psychiatry 2011 Apr;82(4):355.

3.

Reekers JA, Lee MJ, Belli AM, Barkhof F. Cardiovascular and Interventional Radiological Society of Europe commentary on the treatment of chronic cerebrospinal venous insufficiency. Cardiovasc Intervent Radiol. 2011 Feb;34(1):1–2. Epub 2010 Dec 7.

4.

Ameri A, Bousser MG. Cerebral venous thrombosis. Neurol Clin. 1992 Feb;10(1):87–111.

5.

Lim CL, Keshava SN, Lea M. Anatomical variations of the internal jugular veins and their relationship to the carotid arteries: a CT evaluation. Australas Radiol. 2006 Aug;50(4):314–18.

6.

Alper F, Kantarci M, Dane S, Gumustekin K, Onbas O, Durur I. Importance of anatomical asymmetries of transverse sinuses: an MR venographic study. Cerebrovasc Dis. 2004;18(3):236–9. Epub 2004 Jul 23.

7.

Surendrababu NR, Subathira, Livingstone RS. Variations in the cerebral venous anatomy and pitfalls in the diagnosis of cerebral venous sinus thrombosis: low field MR experience. Indian J Med Sci. 2006 Apr;60(4):135– 42. Widjaja E, Griffiths PD. Intracranial MR venography in children: normal anatomy and variations. AJNR Am J Neuroradiol. 2004 Oct;25(9):1557–62.

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Dowson AJ. Analysis of the patients attending a specialist UK headache clinic over a 3-year period. Headache 2003;43:14–18.

10. Guerrero ÁL, Rojo E, Herrero S, Neri MJ, Bautista L, Peñas ML, et al. Characteristics of the first 1000 headaches in an outpatient headache clinic registry. Headache 2011 Feb;51(2):226–31. 11. Tsushima Y, Endo K. MR imaging in the evaluation of chronic or recurrent headache. Radiology 2005 May;235(2):575–9. 12. Good CD, Johnsrude IS, Ashburner J, Henson RN, Friston KJ, Frackowiak RS. A voxel-based morphometric study of ageing in 465 normal adult human brains. Neuroimage 2001 Jul;14(1 Pt 1):21–36. 13. Katzman GL, Dagher AP, Patronas NJ. Incidental findings on brain magnetic resonance imaging from 1000 asymptomatic volunteers. JAMA 1999 Jul 7;282(1):36–9. 14. Morris Z, Whiteley WN, Longstreth WT Jr, Weber F, Lee YC, Tsushima Y, et al. Incidental findings on brain magnetic resonance imaging: systematic review and meta-analysis. BMJ 2009 Aug 17;339:b3016. doi: 10.1136/bmj.b3016. 15. Ayanzen RH, Bird CR, Keller PJ, McCully FJ, Theobald MR, Heiserman JE. Cerebral MR venography: normal anatomy and potential diagnostic pitfalls. AJNR Am J Neuroradiol. 2000 Jan;21(1):74–8. 16. Rao S, Higgins JNP. What is a normal magnetic resonance venogram? Presented to British Society of Neuroradiologists 2002. 17. Higgins JN, Gillard JH, Owler BK, Harkness K, Pickard JD. MR venography in idiopathic intracranial hypertension: unappreciated and misunderstood. J Neurol Neurosurg Psychiatry 2004 Apr;75(4):621–5. 18. Durgun B, Ilglt ET, Cizmeli MO, Atasever A. Evaluation by angiography of the lateral dominance of the drainage of the dural venous sinuses. Surg Radiol Anat. 1993;15(2):125–30. 19. van der Schaaf IC, Velthuis BK, Gouw A, Rinkel GJ. Venous drainage in perimesencephalic hemorrhage. Stroke 2004 Jul;35(7):1614–18. Epub 2004 May 27. 20. Hentzer E. Objective tinnitus of the vascular type. A follow-up study. Acta oto-laryngologica 1968;66: 273–81.

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Contacts/correspondence Dr Nicholas Higgins, Radiology Box 218, Addenbrooke’s Hospital, Hills Road, Cambridge CB2 0QQ Email: nick.higgins@addenbrookes.nhs.uk

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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CRPS case study: return-to-work rehabilitation using graded motor imagery, exercise and work hardening Deborrah Thornhill MCSP SRP

Abstract This is a case history regarding a 52-year-old male ambulance worker, who was unable to work for 23 months due to increasing pain in his right ankle and neck; despite surgery, symptoms continued to progress, a diagnosis of complex regional pain syndrome (CRPS) had been made by his surgeon and ill-health retirement was being considered by his employers. The treatment method used was ten sessions of occupational health physiotherapy comprising a bio-psychosocial, cognitive behavioural approach to rehabilitation, graded motor imagery, graded exercise and a phased return to work. Results: Mr C made considerable physical improvements, his symptoms reduced and he was able to return to full duties in a heavy-demand role as an ambulance worker. Conclusion: The results of this case study suggest that this method does give results that reduce pain, suffering and costs to organisations. Further case studies are required; if they continue to provide good results a randomised controlled trial (RCT) to test this treatment method may be warranted. Keywords Complex regional pain syndrome (CRPS), CBT, chronic pain, central sensitisation, graded motor imagery and rehabilitation, graded return to work

Introduction History of condition Mr C had shoulder surgery in 2008 to remove a ‘lump’; he reports good recovery and successfully returned to work on full duties. In February 2009 he injured his neck after slipping at work on ice; he had several courses of manual physiotherapy, but did not recover. Neck pain and stiffness persisted and the physiotherapist referred him to his GP for further investigations. In August 2010 Mr C found a lump in his foot which made walking painful. Symptoms worsened and in October 2010 he was sent home from work due to intense pain in the right foot and neck; he remained off work until June 2012. In March 2011 he had surgery to remove a ganglion and debride the peroneal tendon in the right foot. Mr C was placed in a cast for six weeks. Mr C experienced problems with his post-op rehabilitation of pain, reduced mobility and ankle swelling, which persisted after the cast was removed. Six weeks later, he was referred for neck surgery. Mr C reports he was told that ‘there were brittle bones in his neck which had possibly broken’. He had a cervical discectomy and fusion at C5/6 level. Mr C was discharged from hospital with a soft collar and he reports that he was not given Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


adequate pain relief, was not made aware of what to expect following surgery or how and when he would return to work. Several months later, Mr C was taking tramadol, paracetamol, gabapentin and solpadol to manage his pain but was ‘trying to do without medication wherever possible’, believing it was unwise to take excessive medication. At the initial rehabilitation assessment in March 2012, Mr C was under the care of two surgeons, a pain clinic and his employers’ occupational health department. He was waiting for an MRI scan to investigate his deteriorating right ankle symptoms. He was worried about the need for further surgery. Initial assessment findings indicated that Mr C had reduced neck and ankle movement, both limited by pain; Mr C was taking inadequate pain medication. The right ankle was discoloured and swollen with areas of numbness, hyperalgesia and allodynia. Mr C had difficulty sleeping; he walked with a marked limp and relied on maximal support from a crutch. Despite pain, Mr C walked daily, believing this would benefit his condition and hasten return to work; despite his active approach, symptoms were worsening. He was frustrated because he did not have a diagnosis for his pain and was not improving, he was concerned about ill-health retirement and confused about the nature of his condition and how best to manage his symptoms.

Bio-psycho-social assessment findings: Thoughts/beliefs Mr C was worried about why his problems had not improved and thought there were a number of factors that may be contributing to his problem, including: deep vein thrombosis, over-tightened tendons, infection, unhealed tissues and insufficient post-operative physiotherapy. Despite his situation, Mr C remained positive and reported ‘it will get sorted, but it is taking a very long time and it is annoying’.1

Behaviour Mr C had been in contact with both surgeons to try to understand the cause of his problems. He had also contacted his MP, reporting: ‘I want him to investigate the NHS’s failure to deliver every step of the way.’ Despite significant pain, Mr C continued to perform light household tasks and walk six miles a day using his crutch He reported ‘it’s mind over matter, I try to get on with life, I don’t want to be sat around moping’ and ‘I try to do normal things to increase my blood circulation but I can’t do much because it’s painful’.

Feelings Mr C was confused, anxious, frustrated, depressed and worried. He had not got a satisfactory answer to his questions or solution to his pain problems. He reported: ‘I am frustrated at being messed about and not being able to get to work’; ‘I want this sorting, I want to get back to work, I am fed up – out of my head – it’s unreal and it’s driving my wife around the bend, she can only put up with me for short periods of time.’2 The threat of ill-health retirement and an increasingly strained relationship with his wife were increasing his fear and anxiety.

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Symptoms and findings at initial assessment 2.5 hours Over the previous nine months Mr C reported that his ankle pain had intensified and spread from the scar to a wider area measuring 20.5 x 15 cm spreading over the outside of the ankle. Mr C was unable to tolerate any touch or pressure, including bedclothes on his foot. He reported a burning pain and was unable to fully weight bear on the right foot; there was numbness in the tips of the toes on the right foot. His ankle and foot circumference was 2 cm greater than the left due to the swelling. Colour changes were evident: the right foot was red and purple. Ankle and neck movement were severely restricted, and limited by pain. Measures are detailed in Table 1 below. Questionnaires completed during the assessment indicated that Mr C was fearful of work tasks, scoring 35 on the Fear Avoidance Beliefs Questionnaire (FABQ)3; he was depressed and anxious, scoring 17 on the Patient Health Questionnaire (PHQ9)4 and 16 on the Generalised Anxiety Disorder (GAD 7).5 His score of 50% on the Oswestry Neck Disability index (ODI)6, and 39 on the Lower Extremity Functional Scale (LEFS)7 indicated severe disability (see Table 1). The clinical diagnosis was complex regional pain syndrome (CRPS); Mr C had altered pain mechanisms due to neurological changes both centrally and peripherally.8 This was the most likely cause of his worsening sensory symptoms in the right foot, neck pain and stiffness. Mr C was not taking sufficient pain medication. He demonstrated ‘boom and bust’ behaviour, for example walking six miles every day and continuing to perform household tasks despite strong pain. He had a poor understanding about both conditions, believing musculo-skeletal structures were at fault and the root cause of his pain. His failure to improve, lack of a diagnosis and effective treatment resulted in low mood, frustration and anxiety with regard to work, the future and his personal relationships. Mr C reported that he was feeling desperate and ready to commit to anything that would help him get back to work. His goals included: return to work on full duties, return to gardening and to lose 2 stone in weight,9 which he reported he had gained over the course of his problem.

Method Mr C attended ten one-to-one sessions over a 16-week period. Session times ranged between 30 minutes and 2 hours. Sessions 2, 3 and 4 included education about pain mechanisms. This was provided in a variety of ways, including hand-drawn diagrams, PowerPoint presentations, discussions and provision of reading material.10 A range of explanations were given to help him understand more about his condition. His body, brain and nervous system were compared to a house fitted with a computerised burglar alarm system which had multiple sensors. This had become mis-wired, and at the slightest movement, the alarm would sound. Movement in the house was equivalent to mechanical stress such as walking, feeling pressure from the bedclothes or from wearing shoes; this was causing Mr C’s sensitised nervous system and brain to produce pain in the foot and neck.11 Having understood this he realised that continuing to walk was unhelpful and likely to increase the alarms in his system. He was happy to replace walking with cycling on a static cycle for up to 1 hour at a time. This was pain-free; a good way of expending energy which would assist with weight loss, improved mood and the endorphin release would also help to reduce pain.12,13 Mr C was able to see how taking pain medication could positively affect his condition and he started to take his medications regularly.

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Non-physical exercises Graded motor imagery: laterality recognition Mr C was unable to move his foot and ankle without increasing pain to level 9/10. In an effort to exercise the motor pathways and remain within pain-tolerant levels, Mr C was provided with a set of 40 cards each displaying a photograph of either a left or right foot held in different postures. Mr C was instructed to identify the laterality of each foot displayed on each card. He was also taught to play card games, including picture pairs, snap and tic tac toe. He was advised that this activity would help rewire his brain and he should do these activities for 20 minutes three times per day. Later he was requested to imagine his own foot adopting the position and later still he actually adopted the position with his foot.14

Mirror therapy Mr C was asked to get a chevalier mirror to carry out mirror therapy exercises at home. He was shown how to place the left foot in front of the mirror with the right hidden behind. He was taught to look at the reflection of his left foot in the mirror. The reflected ‘right foot’ would be ‘adopted by his brain’ and while moving his left foot, his brain would be ‘tricked and believe that this was the right foot moving – painlessly’. Mr C was advised to carry out this activity for 20 minutes up to three times per day. Later, he progressed the exercise by moving the right foot while it was behind the mirror, remaining in a pain-tolerant zone.15

Physical exercises Mr C carried out a series of endurance exercises in comfortable positions to improve neck movement and to reduce pain. The number of comfortable repetitions was measured. Mr C was asked not to exceed the limit when doing these exercises at home.16,17 Over time, exercises were progressed by increasing the repetitions and adopting antigravity functional postures. By session 4, Mr C was carrying out the recommended non-physical and physical exercises for up to 5 hours per day. By session 5, his foot and ankle symptoms had considerably improved, he was able to wear a normal shoe and had returned to walking short distances without the use of a crutch. Neck mobility and pain had also improved. In sessions 5, 6 and 7, he continued with cards and mirror work, commenced ankle mobility and strengthening exercises and gradually increased functional activities. His walking distance increased from 122 steps to quarter of a mile. Return-to-work activities included practising resuscitation techniques, wearing work safety shoes, lifting weight and walking with weight. Mr C used a diary to monitor symptoms and increase his activities gradually. By session 7 the ankle swelling and pain was minimal; there was no hypersensitivity or allodynia. Neck movement was approaching normal values. During sessions 8, 9 and 10 rehabilitation included work-hardening tasks: walking up to half a mile, climbing up and down stairs while carrying 30 kg weight, lifting up to 45 kg from the floor, and walking with a loaded response bag while carrying a defibrillator. Mr C was fit enough to attend manual handling training sessions and start a graded return-to-work programme progressing from light to full duties. Mr C made a plan to maintain his fitness levels by joining a gym, with support from a

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personal trainer.18 He set his future goals as returning to gardening and DIY tasks, and preventing a flare-up. He was discharged from treatment.

Results Table 1

Measures Area of ankle hypersensitivity when touched Ankle swelling 1–5 Metatarsal heads Left = 28 cm Calcaneum/malleoli L = 37.5 Neck flexion Neck extension Neck side flexion L Neck side flexion R Ankle dorsiflexion L = +5º Ankle plantarflexion L = 65º Pain level where 10 is the worst pain imaginable Colour

Sessions 1–2 20.5 x 15 cm

Sessions 3–4

Sessions 5–6 9 x 5 cm

Sessions 7–8 0 cm

Sessions 9–10

29.5 cm 39.5 cm

29.5cms 38.5 cm

28 cm 37.5 cm

28 cm 38 cm

28 cm 37.5 cm

39º 3º 19º 18º 0º

54º 16º 22º 24º

54º 21º 31º 31º 5º

55º 22º 21º 37º 5º

65º 33º 21º 37º 5º

45º

50º

50º

21º

0 cm

5–9

3–4

0–3

0–3

0–3

Red purple

Red purple

Pink

Walking

6 miles 1 crutch Pain 9–10

122 steps in 3’ 13” Pain 4–6

Normal L=R ½ mile

Driving

Unable

Minimised as recommend -ed using crutch Unable

Normal L=R ¼ mile

Full

Full

Sleeping

Interrupted

Interrupted

Good

Good

Lifting

Unable

Unable

45 kg from floor

Walking with weight

Unable

Unable

45 kg from floor 13 kg =+ 12 kg in hand for 10 mins

41

Drove 40 miles x 2 to appt Good, able to tolerate sheets 30 kg/45 kg from floor Around house and outside

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Stairs with 30 kg for 3.5 minutes Kneeling to perform CPR Domestic chores

Shoe wear

Questionnaires FABQ ODI TSK LEFS PHQ 9 GAD 7

Unable 10 mins at a time using a crutch Orthopaedic shoe

Unable

Loose shoe

15 kg Able

Able

Able

Able

Able

Able

1 hour hoovering Tighter shoe

Normal

Normal

Work shoe loosely tied

Work shoe normally laced

35 50% 34 39 17 16

8 16% 18 75 3 4

Discussion The medical approach to Mr C’s neck and ankle problem that was adopted by his treating medical professionals focused on identifying and treating tissues at fault. When the surgery to his foot resulted in a worsening of his condition, this was a warning sign of the development of a chronic pain problem. Despite this, medical management was continued with major neck surgery, which resulted in further deterioration of his problems. He was not provided with suitable or sufficient physical rehabilitation after the surgery. The lack of response to medical treatments, unclear and changing diagnosis and overall worsening of his condition caused Mr C significant anxiety. Worry in someone with a sensitised nervous system such as Mr C increases pain.16 The cognitive behavioural approach allowed Mr C to express and address his worries. The non-physical rehabilitation techniques of graded motor imagery and mirror therapy were essential at the start of rehabilitation when Mr C could not exercise his right ankle without increasing his pain. These methods tricked his brain into thinking the right ankle was working without setting off pain alarms. Once this had been achieved, the swelling, discolouration and high levels of pain had quickly resolved. At this point, right foot and ankle movements could be reintroduced gradually and a more normal course of rehabilitation was possible with progressive exercises and the reintroduction of functional and work activities.

Cost of treatment-use facts and figures from the case study Absence for 23 months (assumption: six months’ full pay, six months’ half pay, then no pay) £28,512. Cost of physiotherapy £180/day x 10 = £1800. Absence research indicates that this gentleman had only a 10% chance of returning to work after 23 months’ absence. Discussions were in place about ill-health retirement. His condition was gradually deteriorating. The physiotherapy intervention stopped the deterioration and resulted in an improvement such that he was able to return to work on full duties. He has now been back at work on full duties for eight months with no further periods of absence. Without this the organisation would have had to meet the 42

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ongoing costs of work absence at half pay of £1200 per month up to two years and the cost of ill-health retirement. In 2001, this was calculated at £10,000 per year up to normal retirement age.19 For Mr C this would amount to £80,000.

Conclusion This case demonstrates the benefits of a cognitive behavioural bio-psychosocial approach to rehabilitation, along with the wide range of strategies that can be utilised by a physiotherapist, to achieve full fitness and function for a person with long-term pain problems. Medical professionals need to recognise when a medical model is not working early on, and to adopt a different pain paradigm. This would result in better use of NHS funding and would reduce the suffering of a person with a persistent pain problem. The treatments used in this case study are based on current physiological principles, which have not yet been put to the test in randomised controlled trials (RCTs). This case study indicates that this approach can be effective and if we continue to get good results such as this we would recommend that an RCT should be performed to put this to the test. Acknowledgements

Thanks go to Nicola Hunter for reviewing this article. Conflict of interest disclosures

None declared.

References 1

Stroud, M. W., Thorn, B. E., Jensen, M. P., & Boothby, J. L. The relation between pain beliefs, negative thoughts and psychosocial functioning in chronic pain patients. Pain 2000, 84:347–352.

2

Eccleston, C. & Crombez, G. Worry and chronic pain: A misdirected problem solving model. Pain 2007, 132:233– 236.

3

Waddell, G., Newton, M., Henderson, I,. Somerville, D., & Main, C. J. A Fear-Avoidance Beliefs Questionnaire (FABQ) and the role of fear-avoidance beliefs in chronic low back pain and disability. Pain February 1993, 52(2):157– 168.

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Kroenke, K., Spitzer, R. L., & Williams, J. B. The PHQ-9: validity of a brief depression severity measure. J Gen Intern Med 2001, 16(9):606–613.

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Spitzer, R. L., Kroenke, K., Williams, J. B. et al. A brief measure for assessing generalized anxiety disorder: the GAD-7. Arch Intern Med. 22 May 2006, 166(10):1092–1097.

6

Vernon, H. & Mior, S. The Neck Disability Index: A Study of Reliability and Validity. Journal of Manipulative and Physiological Therapeutics 1991, 14(7):409–415.

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Binkley, J. M., Stratford, P. W., Lott, S. A., & Riddle, D. L. The lower extremity functional scale (LEFS): scale development, measurement properties, and clinical application. Phys Ther 1999, 79:371–383.

8

Maihofner, C., Handwerker, H. O., & Neundorfer. B. et al. Patterns of cortical reorganization in complex regional pain syndrome. Neurology 2003, 61:1707–1715.

9

Locke, E. A. & Latham, G. P. Building a practically useful theory of goal setting and task motivation. American Psychologist 2002, 57(9):705–717.

10

Butler, D. S. & Moseley, G. L. Explain Pain (Adelaide: Noigroup Publications, 2003).

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11

Gallagher, L., McAuley, J., & Moseley, G. L. A randomised controlled trial of using a book of metaphors to

reconceptualise pain and decrease catastrophising in people with chronic pain. Clinical Journal of Pain 2013, 29(1):20–25. 12

Janal, M. N., Colt, E. W., Clark, W. C., & Glusman, M. Pain sensitivity mood and plasma endocrine levels in man following long distance running: Effects of naloxone. Pain 1984, 19(1):13–25.

13

Ernst, C., Olson, A. K., Pinel, J. P. Lam, R.. & Christie, B. R. Antidepressant effects of exercise: Evidence for an adult-neurogenesis hypothesis? Psychiatry Neurosci. March 2006, 31(2):84–92.

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Moseley, G. L. Is successful rehabilitation of complex regional pain syndrome due to sustained attention to the affected limb? Pain 2005, 114:54–61.

15

Moseley, G. L., Gallace, A., & Spence, C.. Is mirror therapy all it is cracked up to be? Current evidence and future directions. Pain 2008, 138:7–10.

16

Torstensen, T., Ljunggren, A., Meen, H. D., Odland, E., Mowinckel, P., & Gijerstam, S. Efficiency and costs of medical exercise therapy, conventional physiotherapy and self-exercise in patients with chronic low back pain. Spine 1998, 23:2616–2624.

17

Garaets, J., Goosens, M., de Groot, I., Bruijn, C., de Bie, R., & Dinant, G. Effectiveness of a graded exercise therapy programme for patients with chronic shoulder complaints. Australian Journal of Physiotherapy 2005, 51:87– 94.

18

Schonstein, E., Kenny, D. T., Keating, J., & Koes, B. W. Work conditioning, work hardening and functional restoration for workers with back and neck pain. The Cochrane Database of Systematic Reviews 2002.

19

Pattani, S., Constantinovici, N., & Williams, S. Who retires early from the NHS because of ill health and what does it cost? A national cross sectional study BMJ 27 January 2001, 322(7280):208–209. PMCID: PMC26586. http://www.ncbi.nlm.nih.gov/pmc/articles/PMC26586/ (last accessed 27 February 2013).

Contacts/correspondence Deborrah Thornhill, Bury Physio Clinic, Maynewater Lane, Bury St Edmunds, Suffolk IP33 2AB E-mail: deborrah.thornhill@buryphysio.co.uk

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this paper. We as the authors of this paper assert and retain legal responsibility for this paper. We fully absolve the Editors and Company of JoOPM of any legal responsibility from the publication of our paper on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Early Treatment in CRPS: A Patient’s Perspective Zoe Branka Holland BA (Hon), Qualified Solicitor

Abstract Complex regional pain syndrome (CRPS) is a highly painful condition, confined to limbs. Often the onset of CRPS commences with a minor injury and can be profoundly debilitating if it becomes a chronic long-term condition. Sometimes poorly understood, the opportunity for timely diagnosis and thus early intervention and treatment, resulting in significant improvement of symptoms, can frequently be lost. This is my personal experience of the success of early treatment in CRPS and a brief discussion of my experience of differing approaches to early referral and recognition within the medical profession. Keywords Complex regional pain syndrome, CRPS, steroids, pain management, Bier Bloc, Pamidronate infusion, physiotherapy

Introduction This article is written from a patient’s perspective. I am a solicitor, with no medical qualification. The article is intended to give a personal account of CRPS by way of an individual case study.

Chronology of symptoms and my experience Below is a chronology of events, including a description of clinical symptoms, my experience, treatment and outcome. Notable previous medical history § Migraine sufferer § Shingles in preceding 12 months January 2012 § 1 January 2012: I suffer fall at home. Injury to right dominant arm. I attend A&E. Diagnosis: wrist fracture/scaphoid injury; put in temporary wrist cast. § Three days post A&E attendance, patient returns and plaster cast applied below elbow. § Five days post initial injury, severe burning pain commences. I reattend A&E because pain is increasing and it is worse than initial injury pain. Swelling occurring along forearm to elbow and in hand. I am concerned that cast might be too tight. Reassured and told to attend orthopaedic clinic the following week. Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


§ I attend orthopaedic clinic. A discussion re burning pain in forearm and hand. I was reassured that the pain was associated with wrist/arm injury. Decision made to leave cast in situ for six weeks. § Cast in situ for six weeks. Burning pain continues and pain increases. Mobility of arm decreases. February 2012 § Cast removed in orthopaedic clinic. Discussion with orthopaedic surgeon re burning pain and mottled appearance. Reassured and told to increase movement in arm and wrist. § Orthopaedic review: I enquired whether increasing pain was normal. Again, reassured and physiotherapy is planned. On waiting list for NHS physiotherapy. § Pain in arm continues. Pain and burning pain spreads to full hand, including digits. § Train trip to London for business and, on return journey, the pain becomes unbearable. Difficulty typing because the pain makes me feel nauseous; my arm is now extremely painful. § I research private physiotherapy and an urgent appointment is made. § Sensations of heat and extreme cold commence. Colour change in arm and hand, with pain radiating up towards upper arm. Mottling appearance on occasions. § Heightened sensitivity to cuffs, material and touch. Altered sensation in hand and arm. I notice that when washing face, the palm of the hand feels gritty. § I notice that the affected arm feels larger and heavier than the non-affected arm. § I attend private physiotherapy. I am in significant discomfort and the usage of my arm and hand has become limited. Physiotherapist raises concern. Considers possible early CRPS. Agreed that urgent visits and gentle treatment are required. I am advised to discuss this at orthopaedic appointment. § Physiotherapist recommends self-management, massage, limb association and gentle usage. § End of February: I attend orthopaedic review. Discussion about early stage CRPS. I am told by orthopaedic surgeon that it was too early post injury to have CRPS. Advised that onset of CRPS not seen until at least 12 months. Orthopaedic surgeon questions the physiotherapist’s assessment and judgement. I am referred for an MRI scan to check scaphoid and confirms unwilling to increase analgesia for me. § Physiotherapist writes to my GP recommending the need for medication for pain and referral for pain-management assessment given orthopaedic approach. § I commence Gabapentin and Tramadol prescribed by GP. Pain and autonomic symptoms continue. § Pain-management referral made by GP. I am informed of the waiting list at my local hospital. § I research CRPS and find a treatment programme offered at the Walton Centre. I make contact with Andreas Goebel. Initial advice taken and urgent referral supported. § GP makes urgent referral to Andreas Goebel (Pain-Management Specialist) and appointment given within five days. Suspicion of early stage CRPS. § I continue with private physiotherapy. I am recommended to commence selftherapy by gentle touching of the affected arm and regaining ‘attachment’ to the limb rather than disassociating with the arm because of the pain. § Onset of occasional ‘electric shocks’ felt in the arm causing significant pain, with a pain score of 9/10.

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March 2012 § Early March 2012: I attend the Walton Centre, Liverpool, and I am assessed via Budapest criteria.1 On presentation, I had a median pain score of 7, with pain reaching 9 at its worst. Diagnosis of early stage CRPS. § Treating consultant recommended multidisciplinary approach based on the ‘Four Pillars of Care’2; education about CRPS, self-management involving desensitisation, specialist physiotherapy, steroid therapy, increased pain medication and recommended treatments to include a bisphosphonate treatment (Pamidronate infusion 60 mg) and Bier Block. § Initial treatment plan for medication was to slowly increase Gabapentin to maximum dosage (3600 mg per day), commence use of tricyclic antidepressants (TCAs) by way of Nortriptyline and commence high dosage of steroids prior to embarking on any further treatment. § Recommended treatment plan for medication commenced and supported by GP. Recommended to stop taking Tramadol. § I experienced adverse reactions to Gabapentin, including sedation, confusion and dizziness. These decreased with usage but did not disappear. § No adverse side effects from the steroid therapy. § Specialist physiotherapy continued to encourage increased usage of the arm and hand. Includes ‘desensitisation’ techniques, massage and relaxation. § My pain became more controllable and usage of the arm and hand increased. § Particular difficulty and pain increased when I experienced cold conditions outdoors. Keeping the arm and hand warm with additional clothing helped this. § MRI scan performed via orthopaedic department. Confirmation of no ongoing scaphoid injury or bony injury. § The Walton Centre organises bisphosphonate treatment (Pamidronate infusion 60 mg) to take place early April. § I am unsure about Bier Bloc and I wish to discuss this further with treating consultant at the Walton Centre. April 2012 § Early April: I attend the Walton Centre for bisphosphonate treatment (Pamidronate infusion 60 mg) § Immediate post-infusion period – three days of flu-like symptoms and feeling unwell. § I continue with physiotherapy and usage of arm continues to improve. Pain medication continues but on a reduced dose. § Discussion with treating consultant about Bier Block and a decision is made to proceed. May 2012 § Pain is subsiding; further reduction in pain medication. § Continued ‘desensitisation’ techniques at home and with physiotherapist. § I undergo a Bier Block as a day patient at the Walton Centre. § Some significant immediate relief. This tails off as soon as the Bier Block starts to ‘wear off’ but pain does not return to pre-Bier Block pain level. June 2012 § Significant improvement by end of June. § Pain medication stopped. 47

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§ Enjoyment of warmer weather. First month that the arm and hand does not feel unnaturally cold. January 2013 § I have been left with a residual altered sensation around the wrist area and base of the palm of the hand. § This is localised and manageable with occasional pain-relief medication; pain is triggered by cold temperature and restriction around the wrist such as tight cuffs and bracelets. § At times, I cannot tolerate anyone gripping the arm or hand as this produces an immediate response by way of pain and altered sensation. This, however, is only a temporary response and is easily relieved by pain-relief medication and selfmanagement. § Early treatment (within 12 weeks of onset) was commenced following diagnosis of CRPS using the Budapest criteria.3 On presentation, I had a median pain score of 7, with pain reaching 9 at its worst. I received medication to relieve pain (Gabapentin and Nortriptyline) together with an initial course of steroids, specialist physiotherapy, Pamidronate infusion and a Bier Block. § Symptoms were radically improved within six months of onset. Gabapentin and Nortriptyline were subsequently gradually reduced and then stopped. My use of the arm and hand returned to normal. § I have been left with a residual altered sensation around the wrist area and base of the palm of the hand. This is localised and manageable with occasional pain reliefmedication; pain is triggered by cold temperature and restriction around the wrist such as tight cuffs and bracelets. § At times, I cannot tolerate anyone gripping the arm or hand as this produces an immediate response by way of pain and altered sensation. This, however, is only a temporary response and is easily relieved by pain-relief medication and selfmanagement.

Summary of events § Following my diagnosis of CRPS, early treatment (within 12 weeks of onset) was commenced using the Budapest criteria.4 On presentation, I had a median pain score of 7, with pain reaching 9 at its worst. I received medication to relieve pain (Gabapentin and Nortriptyline) together with an initial course of steroids, specialist physiotherapy, Pamidronate infusion and a Bier Block. § My account illustrates an example of the success of early treatment within the first 12 weeks of onset of symptoms. It also highlights a variance in the approach, treatment and diagnosis to my clinical symptoms within the medical profession. While a treating orthopaedic surgeon dismissed a clinical presentation of CRPS in my right dominant arm and hand, the author was fortunate to receive initial treatment and support from a physiotherapist5 who recognised early symptoms and recommended urgent review by a pain-management specialist. Early intervention and treatment significantly improved my symptoms within six months of onset of clinical symptoms of CRPS in the right dominant hand and arm. The initial onset of burning pain five days post injury and swelling in the cast were more than likely the clinical symptoms of the initial onset of early stage CRPS. These were not recognised via an A&E attendance. Thereafter, the orthopaedic team discounted both the physiotherapist’s and my concerns about the clinical signs of CRPS. The orthopaedic view was that CRPS could 48

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not possibly present so early after an orthopaedic injury. This is in complete contrast to research, which suggests that the onset of symptoms for the majority occurs within one month of the trauma or immobilisation of the limb.6 I am grateful for the perseverance of the treating physiotherapist. Her opinion was that the response to the initial injury had been extreme. There was a possibility that the brachial traction stretch at the time of the fall had resulted in an autonomic response, including altered sensation in the ulnar and median nerve distribution (including hypersensitivity). Within four weeks of onset, temperature and circulatory changes were noted. Pain was significant with disturbed sleep pattern. Significant altered sensation was also present. Timely referral and diagnosis at the Walton Centre, Liverpool, enabled early intervention and treatment, by way of a multidisciplinary approach. Pain-relief medication, bisphosphonate infusion and a Bier Block all contributed to extensive clinical improvement and recovery for me. Significant scientific and clinical advances over recent years have improved an understanding of the causes of CRPS, and support within the medical profession for more effective treatments. This has resulted in the Royal College of Physicians’ ‘Complex Regional Pain Syndrome in Adults: UK Guidelines for diagnosis, referral and management in Primary and Secondary Care’ (May 2012). Having had first-hand experience of CRPS, I applaud the tenacity of those within the specialty of pain medicine who have worked persistently towards building a better understanding of the condition, and who have been responsible for advances in treatments within the NHS. This case study supports the success of early treatment, as well as the UK Guidelines. It also highlights the need for continuing education within the medical profession. Widespread best practice is yet to be achieved. Acknowledgements

Dr Andreas Goebel, Consultant in Pain Medicine, The Walton Centre, Liverpool References 1

Royal College of Physicians. Complex Regional Pain Syndrome in Adults: UK Guidelines for diagnosis, referral and management in Primary and Secondary Care. May 2012:1. Royal College of Physicians. Complex Regional Pain Syndrome in Adults: UK Guidelines for diagnosis, referral and management in Primary and Secondary Care. May 2012:3. 3 Norman Harden R, MD, Bruehl S, PhD, Stanton-Hicks M, MB, BS, DMSc, FRCA, ABPM, Wilson PR, MB, BS. Proposed New Diagnostic Criteria for Complex Regional Pain Syndrome. Pain Medicine 2007;8(4). 4 Norman Harden R, MD, Bruehl S, PhD, Stanton-Hicks M, MB, BS, DMSc, FRCA, ABPM, Wilson PR, MB, BS. Proposed New Diagnostic Criteria for Complex Regional Pain Syndrome. Pain Medicine 2007;8(4) 5 Lindsey Anderson, MCSP, Bridge Physiotherapy, 1 Town Street, Marple, Cheshire, SK6 5AA 6 McBride A, Atkins B. Complex regional pain syndrome. Current Orthopaedics 2005:155–65. 2

Contacts/correspondence Zoe Holland, Managing Director, Zebra Legal Consulting, Eaton Place, 114–118 Washway Road, Sale, Cheshire Email: zoeholland@zebralc.co.uk

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Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

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Clear Effect of Mirror Therapy on Trigeminal Neuralgia Pain Annegret Hagenberg MSc MCSP IASP

1

Abstract Introduction: Trigeminal neuralgia (TN) is one of the most excruciating pain conditions. Mirror therapy (MT) is effective in several chronic pain conditions and with a reversing set-up has been described as effective for facial pain with ongoing benefit five years later. Case presentation: a middle-aged lady was offered MT for her long-standing TN. She experienced pain relief repeatedly and reliably after 10 minutes of massage applied by others and subsequently also on self-application of the massage. However, after several selfapplications, which entailed significant strain of the massaging arm, MT suddenly and completely ceased to show effect. Discussion: The significant and on occasions complete pain relief, albeit only temporary, with no side effects experienced, is promising. Whether the reported pain relief is reproducible in further patients, and whether a spoilt illusion can be restored, needs to be investigated. Conclusion: The reliable pain reduction seen here suggests further use of MT for TN. The benefit of the illusion appears to have been lost through the strain or the vision of the selfadministering arm on the pain-free side of the mirror reflection. Therefore self-administration should be avoided. If this treatment proves effective, a breakthrough in pain relief for chronic unilateral facial pain may be achieved. Keywords laterality, mirror therapy, mirror visual feedback, pain relief, syringomyelia, trigeminal neuralgia

Introduction Trigeminal neuralgia is one of the most excruciating pain conditions and the most common neural malady (1). It is often associated with nerve compression, nerve degeneration or sympathetic encountered in syringomyelia, a central spinal cord syndrome caused by a slowly progressing flute-shaped cavity within the spinal cord(6). Medication used are antiepileptics such as Phenytoin, Carbamazepine (CBZ), Oxcarbazepine (OXC), sometimes complemented with Lamotrigine, or Baclofen (7). Side effects of CBZ and OXC such as sedation, dizziness and gait abnormalities occur frequently, and hyponatremia is a serious risk, for which sodium levels need to be monitored. The dosage is limited by the tolerance of side effects. Lamotrigine is better tolerated but prone to cause skin reactions (7). Surgical procedures bear their own risks. Mirror therapy has been found effective for the treatment of pain in phantom limbs and complex regional pain syndrome (8-12) with a mirror between the limbs; the 1

Research Fellow of the Centre for Medical Humanities University of Leicester

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patient can then see the intact limb and the reflection of it in a place where the affected limb could be. Pain relief through mirror therapy has been well described in the literature over the past 20 years. The neuroscientific mechanisms at work, however, are still subject to much debate. Imaging studies have shown a correlation of cortical changes in relation to pain (13) and a reversal of these changes when phantom limb pain was reduced. The motor cortex, the sensory cortex, and the premotor cortex are involved in these changes (14). Reactivation of the representation area of the affected limb in the sensorimotor cortex has been shown in fMRI studies to correlate with pain reduction after mirror therapy in phantom pain (15) and in plexus avulsion (14). Reversal of dysfunctional cortical changes was reported after mirror therapy (16). Other theories on pain resolution in phantom limbs with mirror therapy are, for example, the mismatch of motor commands with visual and proprioceptive signals overcome with a matching visual input (12, 17), or pain memory replaced by perfect visual input (12). Mirror therapy has also been described as useful in a reversed (or double-reflecting) set-up, and was effective in TN (12 p 1706). Ramachandran and Altschuler referred to Beth Taylor-Schott who had successfully treated her husband’s Anaesthesia Dolorosa pain (a sub-group of TN where the nerve is dissected and pain is experienced in the numb area) with this procedure, weaning him off 3,200mg of Gabapentin completely with ongoing benefit five years later (18, 19). This was confirmed in several personal email communications between the author and Beth Taylor-Schott.

Case presentation A middle-aged lady presented for physiotherapy related to whiplash after a recent car accident. She also complained about long-standing TN on her right side involving the auriculotemporal nerve and the zygomaticofacial area near the ear, and the masseter. The pain was experienced on a daily basis lasting from 1 to 5 hours. On the McGill Pain Questionnaire (20) the pain was described as beating or shooting and hot, and after the pain, exhausting. The pattern was rhythmic, intermittent, and ranged between mild and excruciating. A trigger for the pain was heat, whereas sleep and rest alleviated the pain. The only pain medication used was Carbamazepine and it was taken as needed, which differs from the original recommendation. The evidence was passed on and explained to the patient and she volunteered for a free MT add-on to the whiplash treatment. When the presence of the pain next coincided with an appointment at the practice, two mirrors of 35x45cm with smoothed edges, taped at the back of the interface of the long sides, were placed on the desk in front of the seated patient at an angle of slightly less than 90 degrees, so that the patient could see her reversed mirror images slightly to the right and to the left of the mirror-junction (see fig 1). Given that there are no obvious unilateral marks in the face or hair, the image resembles the usual mirror image of one’s own face, and it can only be recognised as side-reversed with movement or other clues entering the scene. That there were two images either side of the junction did not disturb the patient, as they were identical.

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Figure 1, the set-up of a reversed mirror image

Results On the Numeric Rating Scale (NRS), with 0 being no pain and 10 being the worst imaginable pain (21, 22), the baseline pain before the first treatment was 5/10, burning and tingling. Touch and massage was applied to the patient’s left (unaffected) cheek whilst she was sitting still and watching the procedure in the reversed mirror set-up. After 7 minutes of massaging (as shown in figure 2) she reported a quick sharp pain in her right masseter and she wanted the massage to continue. When prompted to choose between touch, moving skin and massaging the masseter, she indicated massaging the masseter muscle as the best procedure. After 10 minutes and another quick sharp pain, the pain started to decrease and was reduced to 2.5/10 after 15 minutes. She reported this to have been a “very weird” experience; she had never thought looking in a mirror could have any effect on her pain. Interestingly, during the following whiplash treatment she positioned herself on her bad side and was unaware of this all through the treatment, much to her later amazement when recalling this. The TN pain continued to decrease to 2/10.

Figure 2, the procedure: touch on the left side is felt on the left but seen on the right

She took the mirrors home and instructed her husband who applied the massage during three pain exacerbations whilst she was watching the reversed mirror reflection. This reduced the pain from 8 to 2 each time. It always took 10 minutes to

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start to show effect and the effect lasted for about one week each time, which was an immense improvement after having experienced daily pain exacerbations previous to the intervention. To be independent from her husband, she applied the massage with her left hand herself which showed the same effect reliably: each time it took 10 minutes of massaging to initiate the pain decrease which then reliably reduced the pain from high scores to 2/10 or 0/10. The effect, however, did not last for a full week. In addition, massaging her own face for such a long time (about 15-20 minutes) strained her left arm. Remarkably, for a very intense 10/10 flare-up she massaged her left cheek for 15 minutes before the pain started to decrease and, after a 15 min break to relax her arm, she continued to massage for another 10 minutes which reduced the pain to zero for the night. On waking the following morning, the pain was only 1/10 but returned within 15 minutes, peaking at 10/10. Subsequently, she applied another 15 min session which reduced the pain to zero and she stayed pain-free all day. However, after more than 7 weeks of consistently successful mirror therapy, during which time she took Carbamazepine only on very rare occasions, the intervention suddenly ceased to be effective. The pain level could not be changed, no matter how long she massaged and watched, and also massage applied by her husband did not show any effect at all. The pain reappeared as it had been before the intervention was started, with similar severity but reduced frequency (only 3-4 times a week as opposed to every day before the intervention). She took Carbamazepine again accordingly as needed. Two years later the pain suddenly increased and spread, and MRI investigations revealed syringomyelia. The patient never experienced any side effects from MT other than the two quick sharp pains in the affected masseter muscle during the first session. No alterations to the mirror setting have been tried so far.

Discussion It is striking that the pain reduction occurred reliably and repeatedly, and always initiated the reduction after 10 minutes of massaging. Given the fact that trigeminal neuralgia is often a condition of excruciating pain with no easy treatment, this variation of MT is well worth following up. It did not cause side effects or pain increase (apart from the two sharp pain sensations in the affected masseter muscle during the first treatment) and only requires a partner, two mirrors and about 15 minutes time when needed. If this mechanism of pain relief proves transferable to a larger sample and to other pain conditions sensible to touch or tissue pressure, a breakthrough in pain therapy would be achieved. The fact that the patient lay on her bad side after the first treatment without realising raises the question of whether her laterality judgement ability had been affected through the intervention. As the pain decreased during this time, correct laterality perception does not seem to be a prerequisite for successful pain treatment. The fact that the procedure ceased to show effect, suddenly and completely, after repeated self-administration of the massage, suggests that the brain had discovered the illusion into which it had been tricked, i.e., the side being touched is not the side of the pain.

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There are three possible causes for the termination of the pain-relieving effect on self-administration of the massage: 1. During self-application, the patient’s left, massaging arm was reflected as being the right arm, whilst in the peripheral visual field of the patient it was seen as the left arm. This conflicting moving visual clue (the arm) might have spoilt the illusion. 2. The patient complained about the left arm being strained during about 15-20 minutes of massaging. This involved tension in the left trapezoid muscle on the neck with likely overflow to other neck and head muscles, e.g. the sternocleidomastoid muscle and the platysma, and possibly even involving the masseter which was massaged. Proprioceptive feedback from these muscles may have sent augmented laterality information to the brain, causing a detangling of the illusion. 3. On self-application, a slight shift of position of the head might have occurred unrecognised. A position shift spoils the illusion, as the side to which the head shifts or tilts in a reversed mirror set-up is contralateral to the one expected in a mirror reflection. To the author’s knowledge, none of these hypotheses has been researched yet. Studies on metacognition of agency (23) have been conducted, rubber hands are well known to be able to be integrated in one’s own body scheme when seen as being touched whilst feeling the own, hidden hand being touched synchronously (24), even with mismatch of instruments of stimulation and blindfolded (25), but not with incongruent objects of stimulation (dish brush and finger) (26). As the object of stimulation (the cheek) was of the same ‘felt and seen’ nature (though not on the same side), it could be implied that self-administration might be effective, as it was on the first few occasions. Other researchers observed activation in the frontoparietal network during self-attribution of observed movement combined with visual mismatch (27), which might also have been the case in our patient. However, there seems to be no research to date using a reversed mirror set-up investigating the influence of laterality and transcallosal innervation. In stroke, mirror therapy proved successful in a patient with callosal disconnection (28). It would be interesting to try whether the illusion could be restored by laterality training as described by Moseley with hand recognition flash cards (29) or with retraining through intense facial grooming with a normal mirror. If it is possible to unlearn a spoilt illusion, then further benefit might be gained with MT. The patient’s pain medication (carbamazepine) causes tiredness and thus affects her quality of life. Mirror Therapy using a reversed mirror image did not cause any side effects in her or in Beth Taylor-Schott’s husband. During the time when the mirror intervention was effective, she only took Carbamazepine on very few occasions. The patient confirms that the pain has definitely become less frequent and not worse in intensity or duration after the mirror intervention during the following two years before the pain spread and syringomyelia was detected. It is therefore ethically justifiable to continue further use in practice and research when the massage is applied by someone else. However, self-administration of the massage should be avoided as this appears to have caused the ceasing of the effect.

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This case study includes reports given by the patient. Therefore bias is acknowledged. It may be stated that this patient has demonstrated reliability over several years, is not prone to self-delusion from the author’s perspective, and reconfirmed the facts on at least two occasions.

Conclusion Mirror Therapy has shown to be reliably effective for TN pain when the massage was administered by a second person. It suddenly ceased to show effect after several successful self-administrations. It is assumed that the visual illusion had been spoilt by the strain on the administering arm on the same side. Therefore, selfadministration should not be performed. In general, the use of two mirrors, taped on the long sides and moved towards each other’s reflecting side, seems promising in alleviating unilateral facial pain and should be further researched. Written consent from the patient to use the data with a pseudonym for academic and teaching purposes was obtained. Notes about the development in the patient’s condition were recorded during further physiotherapy sessions, and additionally in telephone and face-to-face follow-up conversations. The author takes full responsibility for the content of the case study. There is no conflict of interest. Permission to use the notes for this article was given in writing by the patient. The study received no funding.

Acknowledgements:

Special thanks to the patient for permission to publish her case, to D. and R. Armitage for producing the photos, to J. Aldis for language checking and to colleagues for review.

Statement of conflict of interest: There are no conflicts of interest.

References 1. Punyani SR, Jasuja VR. Trigeminal neuralgia: an insight into the current treatment modalities. Journal of Oral Biology and Craniofacial Research. 2012;2(3):188 -- 97. 2. Graff-Radford SB. Facial pain. Current opinion in neurology. 2000;13(3):291 -- 6. 3. Love S, Coakham HB. Trigeminal neuralgia: pathology and pathogenesis. Brain. 2001;124(12):2347 -- 60. 4. Jackson EM, Bussard GM, Hoard MA, Edlich RF. Trigeminal neuralgia: a diagnostic challenge. The American Journal of Emergency Medicine. 1999;17(6):597 -- 600. 5. Graff-Radford SB. Facial Pain. The Neurologist. 2009;15(4):171 -- 7. 6. Todor DR, Mu HT, Milhorat TH. Pain and syringomyelia: a review. Neurosurgical focus. 2000;8(3):E11 -- 6. 7. Attal N, Cruccu G, Haanpää M, Hansson P, Jensen TS, Nurmikko T, et al. EFNS guidelines on pharmacological treatment of neuropathic pain. European Journal of Neurology. 2006;13(11):1153 -- 69. 8. Chan BL, Witt R, Charrow AP, Magee A, Howard R, Pasquina PF, et al. Mirror therapy for phantom limb pain. New England Journal of Medicine. 2007;357(21):2206 -- 7. 9. Darnall BD. Self-delivered home-based mirror therapy for lower limb phantom pain. American Journal of Physical Medicine & Rehabilitation. 2009;88(1):78 -- 81 10.1097/PHM.0b013e318191105b. 10. Grunert-Pluss N, Hufschmid U, Santschi L, Grunert J. Mirror therapy in hand rehabilitation: a review of the literature, the St Gallen protocol for mirror therapy and evaluation of a case series of 52 patients. British Journal of Hand Therapy. 2008;13(1):4 -- 11. 11. McCabe C. Mirror visual feedback therapy. A practical approach. Journal of Hand Therapy. 2011;24(2):170 - 9. 12. Ramachandran VS, Altschuler EL. The use of visual feedback, in particular mirror visual feedback, in restoring brain function. Brain. 2009;132(7):1693 -- 710. 13. Flor H. Maladaptive plasticity, memory for pain and phantom limb pain: review and suggestions for new therapies. England: Expert Reviews Ltd; 2008: 809 -- 18.

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14. Giraux P, Sirigu A. Illusory movements of the paralyzed limb restore motor cortex activity. NeuroImage. 2003;20 Suppl 1(Journal Article):S107 -- S11. 15. Diers M, Christmann C, Koeppe C, Ruf M, Flor H. Mirrored, imagined and executed movements differentially activate sensorimotor cortex in amputees with and without phantom limb pain. Pain. 2010;149(2):296 -- 304. 16. Foell J, Bekrater-Brodmann R, Diers M, Flor, H. Mirror therapy for phantom limb pain: brain changes and the role of body representation. European Journal of Pain. in press. 17. McCabe CS, Haigh RC, Halligan PW, Blake DR. Simulating sensory-motor incongruence in healthy volunteers: implications for a cortical model of pain. Rheumatology. 2005;44(4):509 -- 16. 18. Taylor-Schott B. USA2008. Available from: http://anadmiracle.blogspot.co.uk/2008/09/ad-miracle-in-news.html. 19. Taylor-Schott B. 2008. Available from: http://anadmiracleongoing.blogspot.co.uk/2008/10/anesthesia-dolorosamiracle-is-ongoing.html. 20. Melzack R. The McGill pain questionnaire: from description to measurement. Anesthesiology. 2005;103(1):199 -202. 21. Ferreira-Valente MA, Pais-Ribeiro JL, Jensen MP. Validity of four pain intensity rating scales. Pain. 2011;152(10):2399 -- 404. 22. Turk DC, Rudy TE, Sorkin BA. Neglected topics in chronic pain treatment outcome studies: determination of success. Pain. 1993;53(1):3 -- 16. 23. Metcalfe J, Greene MJ. Metacognition of agency. Journal of Experimental Psychology: General. 2007;136(2):184 -- 99. 24. Botvinick M, Cohen J. Rubber hands `feel' touch that eyes see. Nature. 1998;391(6669):756-. 25. White RC, Davies AMA, Halleen TJ, Davies M. Tactile expectations and the perception of self-touch: An investigation using the rubber hand paradigm. Consciousness and Cognition. 2010;19(2):505 -- 19. 26. Ehrsson HH, Holmes NP, Passingham RE. Touching a rubber hand: feeling of body ownership is associated with activity in multisensory brain areas. Journal of Neuroscience. 2005;25(45):10564 -- 73. 27. Leube DT, Knoblich G, Erb M, Kircher TTJ. Observing one’s hand become anarchic: an fMRI study of action identification. Consciousness and Cognition. 2003;12(4):597 -- 608. 28. Nojima I, Oga T, Fukuyama H, Kawamata T, Mima T. Mirror visual feedback can induce motor learning in patients with callosal disconnection. Experimental brain research Experimentelle Hirnforschung Experimentation cerebrale. 2013;227(1):79 -- 83. 29. Moseley GL. Graded motor imagery for pathologic pain: a randomized controlled trial. Neurology. 2006;67(12):2129 -- 34.

Contacts/correspondence For Annegret Hagenberg MSc MCSP, IASP, Research Fellow of the Centre for Medical Humanities, University of Leicester, 7 Salisbury Road, Leicester, LE1 7RQ, United Kingdom T: +44 116 229 7605 E: ah413@le.ac.uk

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2013. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

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Estimating the Prevalence of Chronic Pain in a Given Geographical Area i

Martin Seymour MSc FRSPH ii Stephen Patterson PhD MSc DLSHTM

Abstract It is estimated that one in five adults suffers from an appreciable level of pain at any one time. Each year approximately one person in ten develops a form of chronic pain. 67% of patients with chronic pain have musculoskeletal pain. Prevalence estimates, however, vary widely across studies. A pragmatic approach to estimating the prevalence of chronic pain in the population of a Clinical Commissioning Group (CCG) in rural West Suffolk was undertaken by applying the prevalence rates reported in three contemporary studies to the local population. Although these studies were carried out during different time periods and locations, they may be considered to be the best sources we have got to estimate the prevalence of pain in a given population. The prevalence rates reported for the UK ranged from 13% to 50.4% with further within-study variation between sex and age groups, socioeconomic status, locality and other variables. Studies showed an increase in prevalence with age, with the largest number of chronic pain patients in the 45–74-year age band. The studies also report on levels of pain experienced and expressed need of patients, and they point to the disparity between severity and expressed need. Although not perfect, the prevalence of pain derived from applying the study figures gives a rough estimate of pain load in a community while planning for services. Keywords Chronic pain, prevalence

Introduction Chronic pain is defined as pain that persists beyond normal tissue healing time, which is assumed to be three months.1 The available research indicates that around one in five adults is at any one time suffering from an appreciable level of pain, while every year approaching one person in every ten develops a form of chronic pain.2 There is, however, wide variance in prevalence estimates across studies and the availability of robust data on the incidence and prevalence of chronic and other pain in England and elsewhere is limited.3 Published research gives conflicting data resulting from difficulties in finding reliable and valid ways to measure pain, differing clinical definitions and cultural interpretations of pain. The studies considered in this review quote prevalence rates that range from 13% to 50.4% with further withinstudy variation between sex and age groups, socio-economic status and other variables.

i ii

Specialty Registrar in Public Health, Suffolk County Council. Epidemiologist, Suffolk County Council. Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


This article reports on a pragmatic approach to estimating the prevalence of chronic pain by applying published figures to a local population to provide a rough estimate of pain load in a community while planning for services.

Methods A search was undertaken of electronic databases, including Medline, AMed, Cynahl and Psychinfo, and augmented with snowballing and a search of grey literature to identify relevant papers and reports. Lack of consistency between studies in terms of case definition and reporting were limiting factors. Papers also recognised cultural differences both between and within countries in self-reported pain reporting. Three studies reporting on prevalence and related outcomes were selected as being relevant for this review, though they are limited in their generalisability to the West Suffolk population as they were conducted across different populations and healthcare systems in the UK and across Europe. The studies did, however, illustrate the range of prevalence estimates in published papers with a between-study range of 13% to 50.4% for the UK and further within-study variation between age groups, socioeconomic status, locality and other variables. Results from these three studies were applied to the West Suffolk CCGiii population data, to provide an estimate of the pain load in the community which might be of help in planning services. CCG population data was sourced from Open Exeter4 and includes all patients registered with GP practices as at 31 March 2013. The report also drew on findings from related reports including the National Pain Audit.5

Studies selected Table 1 – Selected studies with prevalence rates

Study Elliot et al (1999)6

Cohort Sample drawn from general practices (43% practices) in the Grampian Region; 5036 patients age >25

Health Survey for England (2011)7

Sample of the adult population of England

Breivik et al., (2006)8

46,324 respondents undertaken in 2003 across 15 European countries and Israel

Prevalence of chronic pain Grampian population 50.4% (95% CI 45.35–53.7%) General population 46.5% (95% CI 44.8–48.2%) after age and sex standardisation Men 30.9% (95% CI 29.1–32.6%) Women 37.5% (95% CI 35.9–39.1%) Europe 19% (range 12–30%) UK 13% (95% CI 12–14%)

Two reports, Elliot et al. (1999)6 and the Health Survey for England 2011,7 Volume 1, Chapter 9 provide data from the UK. Elliot et al. (1999)6 reported on data obtained from a cross-sectional survey of pain undertaken in the Grampian region of Scotland in the late 1990s. The Health Survey for England7 data is more recent and was collected from throughout England. A European survey, the ‘Pain in Europe’ survey (Breivik et al., 20068), was undertaken in 15 countries in Europe and Israel and it is based on more than 46,000 interviews. While all three have limitations in their relevance to Suffolk, they do provide some insight into likely prevalence of pain in the community. iii

West Suffolk Clinical Commissioning Group is responsible for commissioning health services for 25 general practices service residents of rural West Suffolk (http://www.westsuffolkccg.nhs.uk/about-us/).

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Results Estimated prevalence of chronic pain Chronic pain is defined within the Health Survey for England (HSE)7 as pain or discomfort that troubles a person all of the time or on and off for more than three months. Questions about pain were asked of all adult participants in the main HSE7 interview. Adult participants were asked if they were currently troubled by pain and discomfort either all of the time or on and off. If participants reported that they were, they were asked further questions about the duration of the pain and, if longer than three months, were asked more detailed questions about their pain or discomfort. These questions included: § which areas of the body the pain was in; § the intensity of their pain; § how much their pain interfered with aspects of their life; and § support or help they had received. Overall, around 31% of men and 37% of women taking part in the HSE reported chronic pain. The prevalence of chronic pain increased with age, with older people being more likely to report chronic pain than younger people. In those aged 16–34, 14% of men and 18% of women reported chronic pain. This rose to 53% of men and 59% of women aged 75 and over. Table 2 shows the number of residents aged 15 and over within the West Suffolk CCG population estimated to be experiencing chronic pain based on the age and specific prevalence figures from the HSE7 report. Table 2 – Prevalence of chronic pain in West Suffolk by age and sex estimated from prevalence data based 7 on the HSE report (adults aged 15 and over)

By age and sex

Total

West Suffolk CCG population

HSE, 2011. prevalence %

Estimated West Suffolk CCG patients (95% CIs, Wilson method9)

Male

Female

Male

Female

Male

Female

Total

98,493

101,869

30.9

37.5

30,434

38,201

68,224

(28,661– 32,109)

(36,571– 39,831)

(65,232– 71,939)

Age 15–34

26,855

26,322

14

18

3,760

4,738

8,498

35–44

16,098

15,599

30

31

4,829

4,836

9,665

45–54

17,497

17,309

33

42

5,774

7,270

13,044

55–64

14,817

15,522

43

51

6,371

7,916

14,288

65–74 75+

13,643

14,434

48

55

6,549

7,939

14,487

9,583

12,683

53

59

5,079

7,483

12,562

Elliot et al. (1999)6 in their study of the epidemiology of pain in the community (the Grampian study) aimed to quantify and describe the prevalence and distribution of chronic pain in the community. A cross-sectional survey was undertaken with a random sample of 5,036 patients aged 25 and over drawn from the practice populations of 29 general practices in the Grampian region of the UK. 3,605 questionnaires were returned – a response rate of 71.5%. Chronic pain cases were 60

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identified through two questions: one to assess whether pain or discomfort was present, and a second to establish whether this had started more than three months ago. 50.4% of respondents, equivalent to 46.5% of the general population (after age and sex standardisation) self-reported chronic pain. Table 3 applies the unadjusted prevalence rate to the West Suffolk CCG population aged 25 plus. The total number of patients with chronic pain is higher than that estimated using the HSE prevalence rate above and would be higher still if applied to the same age range of 16 plus. This, perhaps, reflects a limitation of self-report data. Table 3 – Prevalence of chronic pain in West Suffolk based on unadjusted prevalence estimates from Elliott 6 et al., 1999

% with Chronic pain (95% CIs)

West West Suffolk chronic pain Suffolk (95% CIs, Wilson population MethodError! Bookmark not defined.)

Male

48.9 (46.4, 51.4) 85,602

41,859 (39,707, 44,017)

Female

51.8 (49.4, 54.2) 89,372

46,295 (44,112, 48,470)

Age group

25–34

31.7 (27.8, 35.9) 27,789

8,809 (7,723, 9,972)

35–44

41.2 (37.1, 45.4) 31,697

13,059 (11,773, 14,384)

45–54

50.5 (46.4, 54.6) 34,806

17,577 (16,142, 19,010)

55–64

57.1 (53.0, 61.1) 30,339

17,324 (16,077, 18,541)

65–74

57.3 (53.1, 61.4) 28,077

16,088 (14,910, 17,238)

75+

62 (57.1, 66.6)

22,266

13,805 (12,724, 14,835)

Total

50.4

17,4974

88,187 (79,349, 93,979)

Breivik et al. (2006)8 undertook a survey of chronic pain in Europe (the Pain in Europe (PIE) survey) to better understand prevalence, impact on daily life and treatment. This was a large-scale computer-assisted telephone survey of 46,324 respondents (54% response rate) across 15 European countries and Israel in 2003. The study aimed to explore the prevalence, severity, treatment and impact of chronic pain. Chronic pain was defined as pain suffered for longer than or equal to six months, with occurrence in the last month and several times during the last week; a more stringent definition than that applied to the two studies above. The survey also questioned pain intensity on a scale of one to ten. Results show an estimated prevalence of one in five (19%) adults aged 25 years plus with persistent moderate to severe pain of more than six months’ duration. Prevalence ranged from 12% (Spain) to 30% (Norway), and 13% in the UK. The PIE survey has limitations in its generalisability to West Suffolk insofar as it shows wide variation between and within countries in measures of prevalence, consequences and treatment. Of the 3,800 respondents to this survey from the UK, 13% were found to be suffering from persistent pain as defined, which was below the overall average observed in this study of 19%, itself below the levels identified in the HSE7 and by Elliot et al.6

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Table 4 applies the prevalence rates for Europe and the UK from the PIE survey to the West Suffolk CCG population. Table 4 – The prevalence of chronic pain in Europe. Breivik H et al. Survey of Chronic Pain in Europe: 8 Prevalence, impact on daily life and treatment. European Journal of Pain 2006;10:287–333

Adults 18+

% with chronic pain (95% CI)

Europe-­‐wide 19 (18.7, 19.4) UK 13 (12, 14)

West Suffolk population 187,281 187,281

West Suffolk chronic pain (95% CI, Wilson method9) 35,583 (34,920, 36,257) 24,347 (22,414, 26,419)

While recognising the variation in case definition and age range, the three studies provide estimates of the adult population in the catchment of West Suffolk Hospital of between 24,347 (13%) (PIE UK prevalence estimate) to 88,187 (46.5%) (Grampian study), with the HSE analysis providing a mid-range estimate of 68,224 or 34% of the adult population. The three studies provide range of pain prevalence in the UK from 13% to 50.4%, somewhat limiting the utility of the estimates.

Prevalence projections to 2020 It is also standard practice to use published figures from other studies to estimate the projected pain load over a period of time, although these figures need to be treated with great caution. The projection only reflects the population aging and no other factors. It is always likely that various potential interventions including lifestyle changes, advances in therapy and other unknown factors could influence the projected pain morbidity in a given community. At the time of writing, no population projections had been produced for the West Suffolk CCG population. There are no statutory geographies for CCGs and current boundaries may be subject to change. Population projections to 2020 using Office of National Statistics (ONS) and Public Health England data have, however, been calculated for the catchment area of the West Suffolk Hospital in Bury St Edmunds. This catchment population is broadly similar to the CCG population. The percentage increase in the West Suffolk Hospital population between 2010 and 2015 and 2015 and 2020 has been applied to the current CCG population to provide an estimate of the expected population growth in the current CCG area. Table 5 provides an estimate of the projected prevalence based on these West Suffolk CCG population projections and the HSE7 prevalence data. The calculations assume no increase in the prevalence of chronic pain over time with the number of patients with chronic pain increasing in proportion with the increasing adult population. Table 5 – Projected prevalence of chronic pain in West Suffolk CCG

2010 2015 62

West Suffolk CCG population

HSE, 2011. prevalence %

Male 98,493 103,122

Male % 31 31

Female 101,869 102,480

Estimated WS CCG patients with chronic pain

Female % Male Female 37 30,533 37,692 37 31,968 37,918

Total 68,224 69,886

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2020

107,453

107,194

31

37

33,311 39,662

72,972

Population projections for the catchment area of WSH based on data produced by Erpho and ONS, 2010 Prevalence data from Health Survey for England, 2011, Volume 1 Chapter 5. Assumes no increase in prevalence of chronic pain.

Level of pain, expressed level of need and impact on activities An estimate of the levels of pain experienced, expressed level of need and impact on daily lives can help inform service planning, recognising that not all people with chronic pain seek help for their condition. The HSE7 and Grampian6 Error! Bookmark not defined.studies provided an estimate on the level of expressed pain within their respective study populations The Grampian study incorporated a seven-item questionnaire that measures the severity of chronic pain in three dimensions of persistence, intensity and disability. The results enabled patients to be classified into one of five hierarchical grades from zero to four (Table 6). The authors also reported on patients’ expressed level of need, measured through a four-question questionnaire that asked about help-seeking behaviours – treatment sought, use of painkillers and frequency of visits to health professionals. The results were presented as five levels of expressed need (Table 7) from 0 = no need, to 4 =high expressed need. The survey showed that of those patients with chronic pain, 17.2% expressed no need and 28% showed the highest level of need. Table 6 – Self-reported pain severity, adapted from Elliott et al., 1999

6

Self-­‐reported chronic pain severity (%)

Grade i

Grade ii

Grade iii

Grade iv

50.2

25.4

10.1

14.3

Female 47.2 Age group 25–34 56.6

23.5

12.1

17.2

25.2

6.9

11.3

35–44 45–54

56.9 50.7

22.7 26.8

8.1 8

12.3 14.5

55–64 65–74 75+

48.2 49.3 32.3

23.4 24.6 23.6

11.7 13.6 17.3

16.7 12.5 26.8

Total

48.7

24.4

11.1

15.8

Sex Male

Table 7 – Expressed need of patients with chronic pain. Percentages are of total with chronic pain in each 6 subgroup. From Elliott et al., 1999. The epidemiology of chronic pain in the community.

Expressed need of patients with chronic pain (%)

63

Male

Level 0 20.4

Level 1 18.5

Level 2 22.9

Level 3 12.9

Level 4 25.2

Female Total

14.3 17.2

13.7 16

26.2 24.7

15.3 14.2

30.5 28

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The report highlighted the disparity between the measures of severity of chronic pain and the expressed need of patients, and suggested this might highlight areas of unmet need and may also highlight patients who display high expressed need yet mild chronic pain. Participants in the HSE7 survey were similarly asked a number of the questions based on the Von Korff Graded Chronic Pain Scale Version 2.0.1210 in order to rate their current level of pain (‘How would you rate your pain right now?’), the level of their usual pain at times when they had been in pain in the last three months and the level of their worst pain in the last three months. All levels were measured on a scale from zero to ten, where zero was no pain and ten was pain as bad as it could be. Further questions established the number of days they had been kept from their usual activities in the last three months, and ratings – again on a scale from zero to ten – about how much their pain interfered with their ability to carry out their daily activities; recreational, social and family activities; and work, including housework. From these questions a characteristic pain-intensity score and a four-item disability score were calculated. Using these two scores, a chronic pain grade was calculated for each participant. Grades 1 and 2 indicate low interference pain at different levels of intensity, while 3 and 4 indicate pain with differing levels of restriction to usual activities. Table 8 is adapted from the HSE 20117 methods and documentation, and indicates the percentage of respondents at each chronic pain grade. 7

Table 8 – Source HSE, 2011 Aged 16 or over with chronic pain

Base Men 16+ Women16+

Characteristics

% (p)

95% CI

Have chronic pain Chronic pain grade Grade i Grade ii Grade iii Grade iv Have chronic pain Chronic pain grade Grade i Grade ii Grade iii Grade iv

30.9 15.1 6.3 3.6 5.5 37.5 14.4 10.8 4.9 7.2

Lower 29.1 13.8 5.5 2.9 4.8 35.9 13.2 9.8 4.3 6.4

Upper 32.6 19.4 7.1 4.3 6.3 39.1 15.5 11.7 5.5 8

Observed and age standardised rates were produced at regional level but these showed no significant variation across areas. The majority of men and women with chronic pain were assigned low interference grades on the scale. The likelihood of having high interference pain increased with age. There is also a social gradient with chronic pain grade with those from the lowest quintile of equivalised household income more likely to have more limiting chronic pain. The HSE7 survey report states that 61% of men and 54% of women (all England rates) with severely limiting chronic pain (grade 4) and 52% of men and 40% of women 64

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with moderately limiting pain (grade 3) reported having seen a professional (described as a doctor, nurse or physiotherapist) at a specialist pain clinic. The HSE report does not, however, provide information on whether this was in a hospital, community or primary-care setting, nor does it indicate the level or type of treatment received. While the three studies provide a useful indication of the severity and intensity of pain and expressed level of need in the study populations, this is of limited value when assessing local health service need. Limitations in respect to differing definitions of pain, cultural factors and the social gradient in chronic pain, together with varying service thresholds and the mix of services that may be available mean that extrapolation from these reports for the purpose of service planning could be misleading. Hence, it is essential that local data collection and analysis is undertaken. The three studies included in this review provide varying estimates of prevalence of chronic pain in the West Suffolk population. Two studies represent extremes of the estimates within the studies identified in the literature; the third approximates the central measure. This variation reflects differences in research methods and definitions of chronic pain together with geographical and cultural differences. The difference between the three studies needs to be recognised. A key difference is the definition of chronic pain adopted by each study. The PIE8 survey adopted a measure of moderate to severe pain identified through a pain-intensity rating scale. Pain must have been present for at least six months’ duration, been experienced in the last month and at least two times per week. The HSE7 asked respondents if they were currently troubled by pain or discomfort and, if they were, whether they had had this pain or discomfort for more than three months. Those that had were asked supplementary questions, including questions about the intensity of pain. However, the definition of chronic pain was pain or discomfort that had troubled the participant all of the time or on and off for more than the last three months. The measure was self-reported. The Grampian6 study similarly defined chronic pain or discomfort that persisted continuously or intermittently for longer than three months. These different definitions were, in part, responsible for the variation in outcome between studies and, while such differences may at first appear unhelpful, they have the advantage of informing service providers of the potential differences in service use if different thresholds of need are employed. Each study was undertaken using a cross-sectional prevalence survey. In the case of the Grampian6 study, this was conducted through a postal self-completion questionnaire distributed to patients drawn from 29 general practices and stratified by age and sex. A 71.5% response rate was reported (3,605/5,036). The PIE8 survey employed computer-assisted telephone surveys and surveyed 46,324 people, including 3,800 from the UK. The refusal rate was 46% overall and 54% in the UK. In the HSE,7 questions on chronic pain were included in the face-to-face computerassisted interviews. Participants were selected using a multi-staged random stratified sample. A total of 8,610 adults aged 16 and over were interviewed with a general response rate of 66%. The numbers responding to the questions on chronic pain are not reported. There is inherent bias in cross-sectional questionnaires and the extent of such selection, recording and interviewer bias varies with the methods used; hence, the differences between methods, response rates and weighting need to be taken into account when comparing the different prevalence rates reported. The PIE8 survey highlighted differences in prevalence rates between populations that existed both between and within countries. The higher prevalence rate in the Grampian6 Error! Bookmark not defined.study may, in part, be associated with 65

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eographical differences in interpretations of and experience of chronic pain (Northern Italy reported a prevalence rate of 32% compared with 22% in the South and a general North/South divide was identified). How transferable the Grampian region data is to the population of West Suffolk is not known. Likewise, how alike the Suffolk population is with the wider UK or England population can only be assumed in the absence of local data. Kirklees has undertaken a local survey of chronic pain as part of its Joint Strategic Needs Assessment.11 This reported that around one in three residents suffered pain problems with more than half of these saying that pain had limited them in some way over the past four weeks. This article reports on a pragmatic approach to estimating the prevalence of chronic pain. It was prepared to provide the background information for a review of pain services in the West Suffolk area. While only three studies were included in this review, a number of alternative published studies were also identified. Where data was comparable, the prevalence rates in these other studies fell within the extremes of the ones included in this report. Our approach has its limitations and, whereas the findings can provide broad estimates of prevalence, the report cannot be considered as robust or reliable as a full systematic review. Nor was this approach intended to be comparable to a healthneeds assessment, which would have provided a more rounded and comprehensive assessment of the prevalence of chronic pain, the health service needs of patients and the views of service users and providers. A local cross-sectional prevalence study would arguably have provided more accurate and locally specific information, particularly in respect to the severity of pain, the impact on daily living and expressed need for services.

Conclusions The report describes a pragmatic approach to estimating the prevalence of chronic pain in the West Suffolk area. It highlights the wide variance in estimates of chronic pain prevalence reported across published studies. This variance is of little surprise given the absence of a consistent definition of chronic pain, the difficulties in finding reliable and valid ways to measure pain and the differing cultural interpretations of pain that are evident in the studies considered in this article. The evidence from the three studies suggests a prevalence of between 13% and 50.4% with variation between age groups and sexes. This equates to between approximately 24,500 and 88,000 adults in the West Suffolk CCG population. These rates are set to grow as a result of the predicted population growth in the catchment area. The aging population will also have an impact on the prevalence figures as pain prevalence increases with age. A health-needs assessment together with the inclusion of a local cross-sectional prevalence survey and service-user consultation would further inform local decisionmaking. It is recognised, however, that such an approach is not always practical within the time and resources available and as such this alternative approach can be of value to planners.

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Acknowledgements

The authors would like to acknowledge the support of members of the West Suffolk CCG and West Suffolk Hospital Chronic Pain Service Model Joint Development Team for their contribution to this report. Thanks also go to Dr Padmanabhan Badrinath for his advice and support in the production of this report. Conflict of interest disclosures

The authors have no conflicts of interest to declare.

References 1

International Association for the Study of Pain. 1986: Pain terms, a current list with definitions and notes on usage. Pain 1986; 24(3):S1–226.

2

International Association for the Study of Pain (2004) Unrelieved pain is a major global healthcare problem. Cited in UCL School of Pharmacy (2012) Relieving persistent pain, improving health outcomes. London. UCL School of Pharmacy and UK Clinical Pharmacy Association.

3

Gill J and Taylor D, 2012. Relieving persistent pain, improving health outcomes. London: UCL School of Pharmacy.

Open Exeter – developed by NHS Connecting for Health. Open Exeter is a web-enabled viewer which provides the facility for CCGs/Agencies to share information held on the NHAIS (Exeter) database with other NHS organisations.

4

5

Price C, Hoggart B, Olukoga O, C De C Williams A, Bottle A. National pain audit, final report 2010 – 2012 London (2012). The British Pain Society, Dr Foster Intelligence and Healthcare Quality Improvement Partnership. Online at http://www.britishpainsociety.org/members_articles_npa_2012.pdf (accessed 03.12.2013).

6

Elliot A, Smith BH, Penny KI, Smith WC, Chambers WA. The epidemiology of chronic pain in the community. Lancet 1999 Oct 9;354(9186):1248–52.

7

Bridges S. Health Survey for England 2011: Vol. 1 Chapter 9: Chronic Pain. Online at https://catalogue.ic.nhs.uk/publications/public-health/surveys/heal-surv-eng-2011/HSE2011-Ch9-Chronic-Pain.pdf (accessed 03.12.2013).

8

Breivik H et al. Survey of Chronic Pain in Europe: Prevalence, impact on daily life and treatment. European Journal of Pain 2006;10:287–333.

9

Association of Public Health Observatories. Commonly used public health statistics and their confidence intervals. March 2008. http://www.apho.org.uk/resource/item.aspx?RID=48457 (accessed 03.12.2013).

10

Von Korff M. Assessment of chronic pain in epidemiologic and health services research: empirical bases and new directions. Chapter in Turk DC, Melzack R (eds). Handbook of pain assessment: third edition. Guilford Press, New York, 2011. Cited in Bridges S (2011) Health Survey for England 2011, Vol. 1 Chapter 9. Online at https://wessexhiecpartnership.org.uk/wires/files/2013/03/HSE2011-Ch9-Chronic-Pain.pdf (accessed 03.12.2013).

11

Kirklees Partnership. Health and Wellbeing: key issues for the people of Kirklees. 2010 JSNA. Online at http://www.kirklees.gov.uk/community/statistics/jsna/jsna.shtml (accessed 03.12.2013).

Contacts/correspondence Martin Seymour, Specialty Registrar in Public Health, Suffolk County Council Email: martin.seymour@nhs.net

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

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Consent Dr Andrew Lawson

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From the Editor: Sadly Andrew Lawson died before his article could be finalised which would have seen it expanded with more examples. It was decided to publish his article in the current form to recognise his work and in memory of him. Keywords Ethics, pain medicine, consent, medical ethics, law

Introduction This is the first of a series of articles about ethics in relation to pain medicine and deals with issues surrounding consent in adults. It’s clear that many doctors get agitated when the issue of consent is discussed. Rather than being complex and seemingly threatening, in effect it is about having good manners, obeying the law and respecting patients. It is a fundamental tenet of medical ethics and law that before any examination, investigation or treatment of a competent patient, consent must be obtained, exactly as in everyday life any one of us needs consent to touch another person. Consent is fundamental to the doctor-patient relationship and rests upon the philosophical principle of patient autonomy - in essence that they make their own decisions. Consent is a patient’s agreement for a health professional to provide care. Consent is also an essential prerequisite to participating in research. Obviously, not all of these activities requires a signature on a form – consent may be given verbally or even nonverbally (the patient holds their arm out for a blood test, for example) – but each requires the same fundamental approach – an explanation and an agreement. Touching a patient without consent may lead to a claim of negligence or battery1. A claim of battery assumes that the patient did not consent, whereas negligence implies that, whilst consent was given, there was either a failure to provide adequate information or misrepresentation. A negligence claim can only succeed if it can be demonstrated that the patient suffered harm. In practice, consent has three purposes. An Ethical purpose - that of respecting a person’s right to self-determination (autonomy), a Clinical purpose - to enlist the patient’s confidence and understanding of the treatments proposed - and a Legal purpose. A health worker who touches a patient intentionally without consent is committing the crime of battery and a tort of trespass and or negligence. To act in a lawful fashion the health worker needs a defence (which the court of appeal has described as a legal flak jacket2), which may be any of the following: the consent of the patient, the consent of another person authorized to give such a consent or the defence of necessity (that is where treatment is deemed essential and cannot wait for consent). Consent must also be valid. It can be considered valid when the patient is competent, the consent has been obtained without coercion or undue influence and the patient has been sufficiently informed. 1

Dr Andrew D Lawson FRCA FANZCA FFPMRCA MSc (Ethics) , Hon Sen. Lecturer, Medical Ethics, Imperial College, MPhil Student (Economic & Social History), Worcester College, Oxford

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It is worth remembering, though, that you only need a flak jacket when people are firing bullets at you! Preventing patients from firing bullets (metaphorical or legal) at you is not just about filling in the consent form, it is probably more about those general issues discussed.

Competency The Mental Capacity Act (MCA 2005) clarifies issues with regard to consent. It clearly states that a doctor should presume a patient is competent, and the burden is on the doctor to demonstrate lack of competence on the balance of probabilities should it come to court. Incapacity (lack of competence) is described as ‘if at the material time he is unable to make a decision for himself in relation to the matter because of an impairment of, or a disturbance in, the functioning of the mind or brain’. To be competent to make a decision the patient must be able to understand the information given, retain that information, integrate the information into the decision- making process and communicate that decision. Doctors are obliged to help patients to become competent if at all possible: the MCA states that ‘all practical steps to help’ must be taken. Furthermore if a patient makes an unwise or potentially harmful decision in the eyes of the attending staff, this of itself cannot be used to determine competency. Impairment of or disturbance in the functioning of the mind or brain may occur in circumstances or conditions such as: •

Mental illness

Significant learning disability

Delirium

Confusion, drowsiness or loss of consciousness from any cause.

Long term brain damage

Concussion or head injury

Alcohol or drug-related effects.

The effect of pain, confusion and medication has recently been discussed in the courts, as such influences are common in hospital patients. In 2004 (EWHC 1279) it was held that only when the patient’s capacity had been ‘completely eroded’ by confusion, shock, pain, fatigue or drugs would consent be negated! To many, myself included, this seems an extreme position. However, that is where we are. Particularly in pain medicine there are no procedures or treatments that cannot wait for potential restoration of ability, unless a procedure is being done to help restore competence. An example of this is the siting of an epidural in an incompetent patient to reduce opioid requirement and facilitate weaning in Intensive Care. Regarding competency, there are two important points to stress. Only the competent adult may consent. There is no doctrine of consent by proxy in English law. A spouse may not consent on behalf of their wife/husband/civil partner, and neither may anybody else. Similarly, when the patient is deemed incompetent (in the absence of an advance directive) treatment may be given in the patient’s best interests and it is for the medical team to decide. It may be useful and polite to discuss the proposed treatment with relatives, but they have no legal standing. A competent adult may, under the MCA, create an enduring power of attorney and the courts may similarly appoint a deputy to make decisions about the incompetent patient.

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‘Sufficiently’ informed? In England and Wales, the law requires that the patient must understand ‘in broad terms the nature of the procedure which is intended’. English law does not require that patients can only be taken to consent if they are aware of all the necessary and

relevant information, the doctrine of ‘informed consent’. Consent is negated when there has been misrepresentation of the nature of treatment or the patient’s condition or where there has been deliberate withholding of relevant information. The consent must be obtained by the operator or someone trained in the procedure. The degree of information required for consent to be valid is commonly accepted to be that of the Bolam test – that of ‘acting in accordance with the practice accepted as proper by a responsible body of medical men’, and this remains the basis for the law in England and Wales. However, in the USA and in the opinion of some Law Lords, the standard of care that the courts will look to is that of the ‘prudent patient’, where the reasonable or prudent patient decides what course of action to adopt. A doctor might be negligent if information was withheld that a prudent patient might wish to know. Where there is a risk, which might have devastating consequences for the patient, then disclosure is indicated. This is true even when such a risk is rare; in an Australian case, a doctor was found negligent for non-disclosure of a risk of blindness when the incidence was 1:14000. In the context of interventional pain medicine, this would mandate the disclosure of the risk of spinal cord damage and death with many spinal interventions. There is little consensus amongst practising doctors as to what constitutes a serious risk that mandates disclosure, and the Department of Health has suggested that patients should be presumed to wish to know all relevant risks3. There may be cases where in the best interest of the patient a risk might not be disclosed, but the courts will make the final decision. If a clinician considers it reasonable to withhold disclosure of risk then that clinician must be able to justify that decision in that particular circumstance. In the clinical setting when considering deviating from disclosure, taking advice from colleagues, hospital ethics committees or hospital legal teams would seem to be sensible, along with proper documentation. One definition of ‘informed consent’ that has been advocated is that:

‘informed consent is that consent which is obtained after the patient has been adequately instructed about the ratio of risk and benefit involved in the procedure as compared to alternative procedures or none at all4’ Coersion or undue influence This is a rare occurrence but cases have been brought in English courts where such influence has been alleged. The key issue is whether a patient’s will is overborne by another’s influence. There is one famous case in English law regarding a young adult Jehovah’s Witness who was deemed to have been pressurised by her mother into refusing a blood transfusion5.

Consent for disclosure Consent is also required for disclosure of information about a patient and this needs to be considered carefully in the context of internet discussion and electronic media. As a general rule patients should be made aware that information about them will in the day-to-day course of events be made available to their healthcare team and administrative support staff. This does not need to be repeated ad nauseam, nor indeed formally documented, but this might be sensible where you feel it could be necessary or worthwhile. That is a judgment call. Anonymised data needs to be just that- anonymised- and can be freely used to discuss a case as long as you are sure that it is not readily obvious about whom you

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are talking. In an emergency, information may be communicated in the patient’s best interests. Information may also be communicated if there is legal sanction, or public interest criteria are met. The patient’s express consent must be obtained before disclosing identifiable information for purposes other than the provision of their care or local clinical audit. There are currently discussions taking place at a national level that are advocating the obtaining of specific consent for data to be used in national audit.

Problems If one sticks to the law and guidelines one cannot really go wrong in a legal sense, but there are some theoretical problems with our notions of consent.

Uncertainty with outcome

The problem of uncertainty poses an obstacle to consent and joint decision-making. It remains a truism that clinicians are often uncertain about what constitutes the best treatment for a given individual. In seeking consent, clinicians are obliged to discuss risks, both of the proposed courses of action and of alternative courses of action. This bombarding of patients with uncertainty has the possibility of undermining clinical effectiveness, for success is sometimes related to confidence in outcomes, as well as overwhelming the patient with information which they may not be in a position to understand or integrate into their decision-making process. In a sense we have made this rod for our own backs by stressing certainty in our training and discussions, and being more likely to discuss benefits of treatments than adverse effects. Medical staff are trained to be positive and patients somewhat expect that! It is a balancing act between paternalism and what has been described as ‘vending machine’ health care, where medical staff take no part in decision-making.

Hidden Paternalism

It has been argued that ‘paternalism exists in medicine to fulfil a need generated by illness’ and that despite attempts to respect autonomy and provide choice, the decision- making process collapses back all too often into a form of paternalistic decision-making. This is often the case and doctors need to be aware that patients may ask them to make such choices for them, and that it may be the appropriate thing to do. Such decision-making, in the best interests of the patient and at their request, is quite acceptable. It would be sensible to document the process when it occurs.

Inferential Component

An educated, reflective, autonomous, informed adult voluntarily consents to surgery. Have they consented to anaesthesia? One of the problems of consent is exactly what the patient is consenting to. It is best to attempt to explain to patients the breadth of consent and ask them to specify anything that they specifically do not consent to (as in blood transfusion with adult Jehovah’s Witnesses).

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Conclusions At the heart of the matter is trust and an ongoing and mutually respectful relationship with patients. The root of the word ‘patient’ is from the Latin for ‘one who suffers’, and pain is perhaps a paradigm example of suffering. We need to be wary of the fact that in some patients suffering produces desperation. A full and honest description of the planned treatment provided in a compassionate and sensitive way is perhaps the easiest way to don a flak jacket. It also represents a portal into inviting patients to take part in the decision-making process of life and health care. Like making a will, it’s perhaps something patients sometimes don’t want to do but it’s an essential aspect of life.

References 1

Sidaway v Bethlem RHG(1985) 1All ER 643 Re W (1992) 4 All ER 627,633 3 DoH (2001c:11) 4 Herring J, Consent to treatment in ‘ Medical Law and Ethics’ OUP 2008 5 Re T (1992) 4 All ER 649 2

Websites http://www.gmcuk.org/guidance/ethical_guidance/consent_guidance_index.asp

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

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‘Experts are forever’ or ‘You only live once’? A Cautionary Tale of Duty and Temptation in Compensation Litigation Mr Julian Benson

1

Introduction I have been inestimably assisted by experts in the widest range of disciplines over 22 years of practice: from handwriting (in a forged-will case), forestry health (in an airliner-crash case), lichen and digital photography (in a tripping case) and enteropathy (in a goat-starving case). More prosaically, I have seen experts of the highest quality assisting the parties and the court in the widest range of medical disciplines, from a failed attempt at penis enlargement, to leg-saving advice from an orthopaedic expert whose e-mail address was ‘traumawarrior@’. This article is not about those experts, or about you (or most of you)! It is about the parameters and pitfalls of providing the court with opinion evidence.

The general part The role of the expert in personal injury litigation may be summarised as: A duty to provide a neutral opinion, informed by relevant expertise, indicating the potential range of opinions in the field, and giving reasons for preferring one’s own opinion. There are other requirements; for example, experts should not take it upon themselves to promote the point of view of the party instructing them or engage in the role of advocates (Civil Justice Council (CJC), 3.2.3). Also, importantly, ‘where there are material facts in dispute experts should express separate opinions on each hypothesis put forward’ (CJC, 3.7.13). As Sir Stephen Sedley explained in the Court of Appeal only last year: … if it was intended here to suggest that the doctor’s role is routinely that of a sleuth, I must express my respectful disagreement. Forensic medical practice has been disfigured in the past by practitioners who took on such a role … (Charnock v Rowan [2012] EWCA Civ 2, 9)

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Recent examples of the ‘sleuth’: a psychiatrist who personally employed an inquiry agent to ‘spy’ on the claimant, or a rheumatologist who made evaluative comments on the financial health of the claimant’s employer company, to promote a thesis that she would have been made redundant but for the accident. An allied failing is the lure of tendention, which is equally easy to expose, and equally harmful to the reception of your opinion. Classic examples are experts who misquote, selectively quote or fail to quote notes or factual evidence contrary to their opinion. It is true that many experts ‘get away with it’ because such a high proportion of cases settle. Some experts seem prepared to ‘roll that dice’ with their reputation – perhaps, unsurprisingly, these experts are often in the twilight of their clinical practice.

So what if an expert tries to ‘help his/her side’? It is trite that professional reputations of all kinds are precious, and can (witness Professor David Southall and Sir Roy Meadow) be lost or tainted in next to no time within criminal or civil litigation. In civil claims, the dangers may be more marked, usually because ‘popular’ experts give evidence time and again for one or other side, who perceive that they will give an opinion which accords with that side’s case. Sometimes, experts simply cannot increase the balance of their instructions because, despite impeccable neutrality, they are ‘seen’ as a ‘claimant’ or ‘defendant’ expert. Such experts ought to be more careful than ever about the neutrality of their opinion.

Is it difficult to be an expert? The expert’s task is – relatively speaking – straightforward:

§ § §

adhere to the clearly stipulated duties; provide an opinion about a subject in which you excel; and receive payment whether a claim is successful or not.

Key difficulties in the litigation context are:

§ §

resisting improper pressures upon your opinion in any given case; and avoiding the temptation to indulge your own peccadillos or ‘amplifiers’.

Resisting pressure It is worth remembering the rule of thumb that your opinion ought to be identical whichever party instructs you. That does not mean refusing to discuss your opinion with your instructing client, or ‘clamming-up’ in the face of suggested amendments to a report, or answers to questions. It does mean drawing the boundary between your role and that of your

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client, and ensuring that your evidence contains your clear, comprehensive, CPRcompliant opinion. Ultimately, it is you, not s/he who will face cross-examination and criticism if you cannot justify your opinion (and/or fail, for example, to take proper account of aspects of the evidence, or acknowledge/explain a range of opinions). Before you sign off any document, have a final reflection whether a fly-on-the-wall judge would feel confidence in the neutrality and expertise it contains.

Peccadillos or amplifiers I was interested by Dr Bass’s comments that injured individuals often bring to bear certain ‘amplifiers’ within litigation. My concern – and experience – is that experts are just as predisposed to bring to bear their own ‘amplifiers’, or perhaps peccadillos. It may seem an obvious point, but experts ought to resist the temptation to make any, let alone every, medical report a crusade to promote a particular approach or opinion – something the court may be persuaded is a ‘hobby-horse’ rather than a fact-specific expert opinion. However, if you do take that approach, or consider that your predilection actually fits with the facts of the case, then state that that is what you are doing, give detailed reasons for your approach and set out the range of opinion more assiduously than ever.

The specific part Two recent personal experiences Peccadillo?

In a case which settled recently, the defendant’s psychiatric expert, Dr Christopher Bass, preferred to describe a claimant’s clinical condition as ‘disproportionate pain and disability following an accident’, commenting that ‘the diagnosis of CRPS Type 1 is controversial and a diagnosis of uncertain reliability’ – despite the specific diagnosis having been made by: § two specialist treating clinicians (Dr Rajesh Munglani and Dr Adnan Al-Kaisy); § the defendant’s own occupational physician, whose clinical examination ‘leaves me in no doubt that [CRPS] is the correct diagnosis’; and § both medico-legal pain experts (Dr Charles Gauci and Dr Jon Valentine). It struck me that, as a psychiatric expert, that expert was unusually strident in his opinions about CRPS, as well as being impervious to any point in the claimant’s favour. The case settled for close to £500,000.

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Trying too hard? In a 2012 case, in the context of a bitter orthopaedic dispute, the defendant’s orthopaedic expert, Mr Ved Goswami, attempted (for the first time in the joint statement) to deal with the absence of pre-accident shoulder symptoms by arguing (and thereby suggesting sufficient expertise to comment) that the claimant’s gout medication masked such symptoms. The court permitted the claimant to rely upon a rheumatologist, Dr Ray Armstrong, whose report explained that the ‘expert’: was asked to produce evidence to support his assertion that allopurinol could have analgesic properties that might mask symptoms of a rotator cuff problem. He has provided no credible evidence to support this position. He has simply extrapolated from the results of laboratory tests on mice to suggest that allopurinol could have analgesic properties in humans. There is no justification for this conclusion at present. I think the fact that [he] relies on this document illustrates the weakness of his position. Once again, the case settled successfully, for £75,000.

Examples of judicial evaluation of expert evidence Just because there are so few reported cases in personal injury practice, the legal community pays great heed to those in which experts have been assessed by the court. In the field of rheumatology and pain medicine, there have been some pointed judicial comments. It is worth noting again how rare it is that cases are contested in court. When they are, judges often eschew direct criticism of experts except in the clearest cases. The following cases might be considered a master class in how to and how not to assist the court. In Balsom v Smith (2008) Northampton County Court, Mr Recorder Clark compared the evidence of two rheumatologists, Dr George Struthers and Dr Robert Bernstein, in a 96-page judgment. The judge commented that Dr Struthers, the claimant’s expert, ‘brought a healthy skepticism to his assessments’ (p. 69) and ‘at every stage I felt completely satisfied regarding his total objectivity, his willingness to reconsider matters in the light of new material and his desire to do his best to assist the court’ (p. 68). There were a host of issues which concerned the Court about Dr Bernstein’s evidence, which the judge stated were ‘not appropriate for the Court to comment’, in relation to ‘the wider implication of Dr Bernstein’s comments on his profession’ (p. 79). Those had included the following:

§

‘He explained that he “bent over backwards” to avoid a diagnosis of Fibromyalgia …’ (p. 76).

§

‘He appeared at one point to cast doubt upon the legitimacy of the labeling of the condition by the American College of Rheumatology (“ACR”) suggesting that there

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may have been professional advantages for rheumatologists if they were able to point to a specific recognized and named condition (a “marketable condition”) as a means of generating work’ (p. 76).

§

In cross-examination, he ‘asserted that the ACR were creating work’ (the judge went on to quote him). ‘It justified rheumatologists getting work … it is a marketable disease so they could have private patients come to their practice’ (p. 78).

§

‘He completed his evidence in chief with an observation that “half of those who come from their doctor with a diagnosis of Fibromyalgia, don’t have it” ’ (p. 78).

The judge referred to those (and other) issues because he was ‘satisfied that they have influenced the manner in which he has approached the case’ (p. 79). Then the judge’s criticisms deepened: It is clear that Dr Bernstein was suspicious of the Claimant before he examined her. Although I am satisfied that he did tell the Claimant that he did not know much about the case, as he acknowledged in cross-examination: ‘I formed a suspicion from what I read – her account was on the bounds of reality before I met her.’ (p. 80) As far as his examination was concerned, he did not obtain the same results from a tender-point test as had other doctors, including Dr Struthers who was ‘surprised that Dr Bernstein had not reached the same conclusion’ (p. 74). As regards the surveillance, when asked to comment on the claimant remaining at home, Dr Bernstein commented that the claimant could be doing ‘what some of my patients term “lying low” ’. When the claimant was observed using a stick or crutches, he preferred to suggest that it was a psychological prop, or ‘an aid to deception’ – indeed the judge found that he did not ‘entertain the possibility the Claimant might have genuine need of such aids’ (p. 81). The judge went on to comment on aspersions cast by Dr Bernstein on the claimant’s solicitors (p. 82) and even upon a treating rheumatologist, Dr Sprya. The judge then went on to comment on evidence which the whole court had heard, and Dr Bernstein’s reaction to it, commenting: I was surprised that, having listened to the consistent accounts given by her family and friends about her decline since the accident, the only point Dr Bernstein relied upon was a comment by Mr Renwick which Dr Bernstein interpreted as possibly undermining the claimant’s case. […] I do not think the evidence was capable of doing so). (p. 84) I think that Dr Bernstein’s approach is illustrated also by his response to the evidence of Iris Steadman (p. 84, resumed on p. 86 after that evidence was reviewed by the judge) ... viewed objectively, that appears to be very

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significant evidence … That Dr Bernstein remained unwilling to attribute significance to this is of concern. (p. 86) Furthermore, ‘Dr Bernstein appeared unnecessarily defensive’ (p.86). The judge ‘found his accentuation of what he considered negative aspects of Ms Balsom’s case, and his unwillingness to acknowledge its positive aspects (including her efforts to get better), to be of concern’ (p. 87). ‘I do not accept the evidence of Dr Bernstein regarding the absence of reaction by the claimant to his examination’ (p. 88). ‘I think it likely that Dr Bernstein had come to a conclusion early on that the Claimant was malingering and/or adopting a sick role’ (p. 88). Perhaps most tellingly of all was the judge’s turn of phrase at the outset of his review of Dr Bernstein’s evidence when he said that he had ‘no doubts regarding his sincerity in pursuing his cause …’ (p. 75) (my italics). In Warren v Jennings (2008) Cardiff County Court, HHJ Seys Llewelyn considered evidence from Professor Jayson (defendant’s rheumatologist) and used a telltale phrase – ‘To put it at its kindest to Professor Jayson, this [the severity of the claimant’s pompholyx at the time of examination] may have led him to a preoccupation with pompholyx as being the predominant or ongoing cause of the symptoms from which the Claimant suffers’ (para. 66). In Connery v PHS [2011] EWHC 1685 (QB) HHJ Platts: ‘Dr Munglani gave his evidence with considerable authority and was impressive’ (para. 48). Dr Webley (rheumatologist) ‘gave much more considered evidence than … Dr Bernstein, who it seemed to me was somewhat skeptical of the diagnosis of CRPS generally, not only in this case, and was striving, without much success, to find some other explanation for the Claimant’s symptoms […] I did not find his reasoning nor his evidence persuasive’ (para. 49) (the judge did not consider Dr Bernstein had been ‘partial’, but then again, he was not shown the judgment in Balsom). I found Mr Boston [defendant’s orthopaedic expert] to be hesitant, and it did not assist my impression of him that he had not read the literature referred to by Dr Munglani before trial, and I was certainly not impressed by the way he purported to be skeptical about it without having considered it (para. 49). ‘Dr El-Assra’s [defendant’s psychiatric expert] opinion [was] extreme’ (para. 51).

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Final word The foregoing are merely examples. There are plenty more in relation to experts in different disciplines (Dr John Searle, within road-traffic reconstruction, for example), to say nothing of adverse judicial comment concerning barristers and other lawyers (no doubt the author included). The simple point of this article is that experts are the bedrock of personal injury claims. If your evidence is not of the highest quality, and neutrality, the system will fail. If you follow the requirements of the rules (while being as robust as you like), you will help to promote earlier resolution of claims, and considerably reduced costs, to the benefit of all concerned. Contacts/correspondence Julian Benson E-mail: julian.benson@guildhallchambers.co.uk

Intellectual property & copyright statement We as the authors of this article retain intellectual property right on the content of this article. We as the authors of this article assert and retain legal responsibility for this article. We fully absolve the editors and company of JoOPM of any legal responsibility from the publication of our article on their website. Copyright 2014. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution and reproduction in any medium, provided the original work is properly cited.

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The Impact of the Jackson Reforms on Medico-Legal Evidence Harriet Formby MA, BA (Hons)

Abstract The recent Jackson reforms to costs in civil litigation have implications for the use of medical experts in personal injury and clinical negligence matters. This is due to an increasing need to justify the use of expert input, given the costs-management focus of the reforms. A study was conducted over the summer of 2013 where 25 firms of lawyers were questioned on their use of experts and their experiences and predictions of the impact of the Jackson reforms on the use of expert evidence. This was conducted through a quantitative online questionnaire supplemented by a series of follow-up, in-depth, qualitative interviews. The findings of the study were that the reforms did have implications for the use of experts. However, little change was anticipated in larger cases, for those experts who adapt well to the changing market needs of lawyers, in response to the reforms regarding case management and costs budgeting. It was recommended that medical experts should increasingly demonstrate pro-activity, justification and accountability concerning the emerging case costs budgeting and management requirements of the reforms. Overall the reforms appear to represent a changing landscape in the Personal Injury and Clinical Negligence market where expert evidence is used, but there is opportunity for forward thinking experts to adapt to meet the new order.

Keywords Jackson reforms, expert evidence, medico-legal

Introduction The study focused on the investigation of the implications of the recent Jackson reforms on the use of quantum accountancy expert evidence in civil litigation. These reforms, known as the ‘Jackson reforms’, come under the Legal Aid, Sentencing and Punishment of Offenders Act 2012 (‘LASPO’), which came into force on 1 April 2013. Justice (2013)1 outlines key changes through these reforms, which have implications for civil litigation. These extend to legal-costs management, proportionality of costs, abolishment of the no-win-no-fee structure, imposition of sanctions to encourage early settlement and (relevant to this study) the use of expert evidence – as per Lord Justice Jackson’s 2010 proposals for costs in civil litigation (Legal World News 2012:596).2 According to Lord Dyson, the Jackson reforms were designed to re-establish ‘equality of arms’ between plaintiffs and defendants in personal injury cases, in order to reduce illegitimate claims and improve defendants’ ability to defend claims (Solicitors Journal 2013:3).3 The Jackson reforms were thus designed to transfer the funding of civil litigation from defendants and their insurers over to claimants and their lawyers. They were intended to improve access to justice and to make costs Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


more proportionate (McIvor 2011:411–13).4 This was in response to the perception that costs of civil litigation were disproportionately high (Crouch 2011:17–19).5 The reforms also tackle case efficiency. Steward and Pratt (2013:23–5)6 suggest the reforms were designed to introduce a more robust approach to case management, in response to perceptions that courts had become too tolerant of delays and noncompliance. There are now tougher sanctions for those who fail to comply with casemanagement directions. The reforms were anticipated to result in a sea change to the funding, budgeting and management of litigation, whereby parties now have to file detailed cost budgets before trials begin and maintain an ongoing focus on proportionality of costs. The reforms had a controversial passage into implementation and were greeted with ‘praise and opprobrium’ (McIvor 2011).7 During the three-month consultation on the proposed reforms, the Ministry of Justice received 600 submissions, mostly in disagreement with Jackson’s recommendations. Notwithstanding opposition, government passed the Jackson reforms. There had been increasing concern about expert witnesses, in practice, being partisan to their clients. According to Lord Woolf (1996),8 this had increased because experts tended to, initially, be instructed to advance the party’s case and then changed roles, to provide evidence suitable for court, where it was then perceived that they may fail to give an independent opinion. According to Solon (2011),9 Jackson LJ stated (at his fourth lecture on the implementation of his Civil Litigation Costs Review) that where the reforms were designed to impact on experts, in essence, this meant, ‘reports should be small but perfectly formed and cost less’. The Jackson reforms introduce new rules regarding the court’s power to restrict the use of expert evidence and the costs involved (Expert Witness 2013).10 Table 1 outlines the new rules of the Jackson reforms that concern expert evidence, which apply on applications for permission to rely on expert evidence made after 1 April 2013. Lord Dyson outlined that as part of the new commitment to proportionality and the fair distribution of court resources, that in some classes of cases, courts may reach decisions based on less evidence than they have had in the past and parties could no longer expect ‘indulgence’ with failures to comply with procedural obligations (Solicitors Journal 2013:3)11. Kennedys (2013)12 suggested an expert’s failure to comply with the court timetable could result in the expert’s fees being disallowed or worse, if there was a consequential delay elsewhere on the case. Regarding experts’ thoughts on Jackson, Solon (2012)13 found that over a third of expert witnesses believe the reforms proposed by Jackson LJ in his 2010 report would lead to injustices (according to a survey of expert witnesses). Table 1 Elements of Jackson affecting expert witnesses, Burn (2013:21– 2)14

Aspect of Jackson reforms CPR Part 35 Rule 35.4(2)

Implication for expert evidence Changes designed to manage costs of expert evidence and to ‘better’ focus experts on the issues in dispute.

Requirement of an estimate of the costs of expert evidence.

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Rule 35.4(3)

The court is now able to specify the issues to be covered by the expert evidence and the type of expertise necessary.

Paragraphs 11.1– 11.4 of PD 35

The court can order experts from like disciplines to give evidence concurrently rather than sequentially as part of ‘their’ party’s evidence (a practice known as ‘hot-tubbing’). This first involves the judge questioning the experts together about disagreements in the joint statement and then by the parties’ advocates.

Methods The study was conducted following a pragmatic research philosophy underpinned by an action research strategy. A mixed-research method was undertaken involving a quantitative online questionnaire designed for solicitors, barristers and judges who dealt with personal injury and clinical negligence cases, and a series of follow-up indepth qualitative interviews. The question design focused primarily on the use of quantum accountancy experts, as a firm of forensic accountants had commissioned the research; however, many of the study findings are applicable to other types of experts also. In particular, at interview, many contributors discussed the reforms in relation to use of medical experts. Contributors The study was conducted in August–September 2013 and had representation from 25 national and large regional firms of solicitors. Contributors were predominately at partnership level and highly specialist. The majority (85%) identified themselves as claimant solicitors, with 15% being defendant solicitors. 65% of contributors identified themselves as having a primary area of interest in personal injury, and 35% in clinical negligence. In addition, a number of barristers and a judge also completed the survey.

Results 1. Changes to use of expert evidence since 1 April 2013 Most contributors had not noticed changes to the use of expert evidence since April 2013, as they were still dealing with cases that had been issued pre 1 April. It was reported that solicitors issued an increased number of claims just before 1 April to delay the effects of Jackson. The feeling was that the industry was in the ‘lull before the storm’ and the question should be asked again in six months’ time. Of those that had noticed changes, while they felt there was little uniformity, anecdotally, they reported more instances of applications for use of expert witnesses being disallowed by court, or their costs limited, and fewer applications for use of experts. There appeared to be general uncertainty and confusion with interpreting the practical implications of Jackson, with suggestions that, due to a lack of training, judges appeared to be just as ignorant as practitioners regarding the implications of the reforms. The odd court, however, was said to be sticking to the rules more stringently. There was a perception that pre-Jackson, judgment had varied between lenient and harsh with a tendency towards either claimant- or defendant-friendly rulings, dependent on the court or location. But there was a feeling that, as long as practitioners were prepared, solicitors would have the same arguments concerning expert evidence after 1 April as before 1 April and perhaps the Jackson reforms would result in a more consistent approach. In general, the ‘horror stories’ seemed to be in the lower-value claims and there were suggestions that if a solicitor was initially told they could not use a particular expert,

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it would probably still be considered again at the next case-management conference (CMC). For high-value work, it was anticipated that, as before, the use of experts would be justified. Frontloading of expert evidence A number of contributors had observed an increase in the ‘frontloading’ of expert evidence, whereby, due to difficulty with justifying the costs of experts at CMCs, solicitors were increasingly instructing experts pre-issue. This was in the belief that if they obtained the evidence before formal court proceedings were issued, it would be harder for judges to subsequently disallow the evidence; although, the frontloading of evidence was said to have been on the increase since the 1999 Woolf reforms. In general, the rate of appointment of experts was expected to remain static for the more complex cases. Some contributors commented that they felt there might be an increase in use of experts in an advisory role, as opposed to as expert witnesses in the future, although this was not expected to change significantly for larger cases. Ability to fight excluded expert evidence There was a perception that it was only a matter of time before lawyers would face excluded expert evidence. Around 60% of respondents stated that if a court determined that they could not use an expert witness, they would use a shadow expert or expert adviser instead. For some contributors this was dependent on the recoverability of shadow-expert fees, either via the client or defendant. A number of contributors felt prepared to fight decisions to exclude expert evidence by gathering more information to justify the use and asking again in the expectation that there would always be an opportunity for a second bite of the cherry. However, challenging disallowed evidence was perceived as risky by some, given the costs associated with appealing. There were suggestions that it would take some ‘brave souls’ to appeal decisions to disallow expert evidence through satellite litigation. It appeared that for cases with ‘suites’ of experts, it was important for defendants to demonstrate that they were not instructing experts to counter all of the claimant’s experts. There was a perception the courts will be more receptive to defendants instructing counter experts only on the most contentious issues. This should mean that experts who deal with contentious issues in complex and high-value cases would still be instructed. Access-to-justice implications 80% of survey participants thought that there were, or maybe, access-to-justice implications to the Jackson reforms. Of those who thought there were, this related to concerns of reduced ambit of instructions and a perception that judges would be keen to limit expert evidence where possible. There was a perception that the motives of the reforms favoured the insurance industry and short-term government costcutting. Those who thought there may be access-to-justice implications felt it was possible in some cases, but that larger cases would escape implications. There was a worry that sometimes the courts only see one end of it (i.e. fast-track or multi-track cases) and some contributors hoped courts would not take an indiscriminate approach to restricting evidence and cap expert fees for the sake of it, under ‘proportionality is king’. There were also concerns that the effects of capped costs may be worse for claimants than defendants. This is due to claimants needing to disclose all costs while defendants can keep some hidden (and treat them as business costs) due to disparities with the disclosure of recoverable costs.

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There were suggestions that some of the potential access-to-justice implications were on the increase pre-Jackson and had been a problem for a while. Contributors cited major issues in fast-track personal injury whereby claimants were ‘processed’ rather than ‘represented’. Some did agree that there had been a need to address proportionality through the reforms, suggesting that lawyers had lost touch with proportionality and there will now be a decrease in ‘frivolous’ claims. Those contributors who felt there was no access-to-justice implications suggested there were enough safeguards in place to ensure justice by appealing any decisions regarding restrictions to use of expert evidence in the right case (such as through the Court of Appeal).This was because they felt judges would be concerned if claimants did not get ‘fair’ compensation. Also, there was the potential to use the expert as a sub-contractor, thereby paying the expert to do the work in place of the solicitor. Some contributors favoured the reforms, believing they would increase competition and create stronger business models going forward, regarding fee structures for experts and solicitors. A number of contributors mentioned justification for reforms, given the high costs of litigated claims. Also, there was discussion, primarily by defendant contributors, of a ‘whiplash epidemic’ of fraudulent claims in lower-value cases, with the idea that insurance premiums would now be reduced due to the reforms. It was felt that this would move the claimant market more in line with the defendant market (lower value) where there were ‘tighter’ costs. Also, there was a feeling that the proportionality focus of Jackson would help stop cases drifting and costs building up, due to more proactive case management. 2. Cost budgets and proportionality The reforms introduced a requirement for cost budgets, which have tough penalties for non-compliance, whereby claimants may only be able to recover court fees. This means there is a need to be very careful, with cost budgets, to get them done on time and allow contingency if anything out of the normal were to happen – as once the budget was approved it was thought that it would be hard to change. Contributors felt a need to approach cost budgets on a worst-case scenario basis (this means that they are coming in very high) and anticipated that they would need to justify use of experts more than before. Solicitors felt cost budgets would require a lot of time/effort and they will use cost draftsmen to produce their cost budgets and to help make the case for expert input. Some defendants said they would pay a premium for what they perceive to be better-quality experts, if this would reduce damages overall. This contrasts with the claimant side appearing to be more price sensitive, due to a need for budget disclosure. There were expectations that, going forward, experts need to become more proactive about costs. A number of respondents predicted that increasingly experts’ costs would need to be identified in advance, for budget requirements. Whereas in the past, under an hourly rate basis, the assessment of experts’ costs tended to be dealt with at the end of the process, costs will increasingly need to be estimated at the beginning of the expert’s involvement and attention paid to the justification of such costs. Of those few contributors who had already been required to submit cost budgets on cases, some felt it had been a ‘filing exercise’, as they had received no comment from the defendants or judge on their submission.

3. Timetabling Almost 80% of survey respondents thought the Jackson reforms would influence when experts were appointed. There were suggestions that it would become increasingly more beneficial to involve an expert earlier on, at the costs-management

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stage, given the need for cost budgeting. Though some felt that the reforms would mean experts would be appointed later, as parties might be more inclined to wait to first see whether courts granted permission for experts. Those who felt experts would still be appointed at the same time as before, felt the reforms wouldn’t affect the timing of the need for expert input, only the recoverability of associated costs. As a result, they thought that the quantification of a claim would progress in the same order as before, particularly if they were not formally appointed as an expert witness at the outset but rather used as a sub-contractor. In light of tough sanctions for non-compliance with cost budgeting and court timetables, solicitors said they would be ‘smarter on timetabling’; some mentioned that they would be doubling or tripling timing on cases to ensure they adhered to court timetables. Some respondents said they were likely to stop using experts who could not comply with stricter timetables. The problem was that good experts were said to be in high demand with long waiting lists, so there was a fear that they would not be able to rely on this evidence. It was suggested that a culture change was needed for experts, regarding costs and report turnaround time. Already some good experts were said to be reducing their workload to ensure they could comply with the stricter timetabling requirements of Jackson; which was felt to be increasingly reducing the ability of solicitors to instruct their preferred expert. There were suggestions that there was no obvious source of the next generation of medical experts. 4. Single joint experts (SJEs) Most contributors felt that instructing SJEs was an impossible position, with 90% of respondents stating they never or rarely used (quantum accountancy) SJEs. There were concerns held of experts’ impartiality and fears of not being able to get permission to instruct another expert if they were not happy with the SJE’s interpretation. Others stated that SJEs only work in the ‘right case’, as often privileged discussions were vital. However, 50% of survey participants did think that using an SJE could be more cost effective than using a separate party-appointed expert. This tended to be based on both parties consequently agreeing on the SJE evidence. Though it was felt that it was unlikely for both parties to agree on this, and if one party sought another point of view, it would then become less cost effective. There was also anticipation that, in many cases, parties would still use shadow experts (and incur the associated costs). Also, some respondents did stress that while it may create savings, this could be at strong risk of not providing the best service to clients. 67% of survey participants agreed that an increase in the use of SJEs was an intended outcome of the Jackson reforms and several of these participants expected more SJEs by court order. However, only 30% of survey participants said they were likely to increase their use of SJEs. Instead, they would argue for sole instruction in the belief that the size of their cases would justify a party-appointed expert. Thus, they would ‘shy away’ from any attempts to impose more SJEs, in some cases, by increasingly ‘frontloading’ expert evidence. Many contributors suspected and hoped that in larger and more significant cases, judges would still take the view for separate experts, if the solicitor had fully justified the use of the expert. 5. Solicitors’ confidence in working within Jackson Almost 65% of contributors felt unprepared for, or unsure about, the Jackson reforms, due to a lack of advance guidance on cost budgeting and the consequences of technical breaches. There were feelings of uncertainty of how the courts would behave and expectations of varied responses dependant on court/judge. Many

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expressed concerns over a general lack of awareness in the industry, of the implications of the rules in practice, based on the lack of practice directions. There were perceptions that the reforms had been ‘rushed through’, so it was felt that until the system was ‘embedded’ through satellite litigation (cases tested at the Court of Appeal/Supreme Court), all would be ‘feeling their way’, particularly as cases were running under two sets of rules (pre-Jackson and post-Jackson). Those contributors that felt prepared for the reforms described having had a large amount of training, webinars and discussions with colleagues over the implications. They tended to be lawyers from larger/national firms who also had systems in place to share data and intelligence and to work out, from their databases of historic cases, what realistic costs should be over different case profiles. 6. Long-term perceptions There was a universal perception that despite the seemingly far-reaching impact of the Jackson reforms, practitioners would eventually revert back to old and established practices, regardless of what the courts try to do – because ‘lawyers are good at thinking ways round things they don’t like’. However, some respondents did not feel this was a realistic view, as they felt there was more intent behind this legislation than previous reforms. These respondents expected no relaxation of the rules beyond iteration, so they emerged as something more workable. There was a feeling that any changes as a consequence of the Jackson reforms were not completely new, as many of the implications had been on the increase before April 2013. Given the view that the implications of the Jackson reforms did not translate well in to day-to-day practice, satellite litigation was seen as inevitable, due to the apparent lack of guidelines on the implementation of the reforms at a practical level. With the courts’ agenda to eliminate experts where possible, it does appear to be a changing landscape for both lawyers and experts. However, it was suggested that for high value, multi-track work there shouldn’t be a particularly different situation, as long as lawyers justified the need for experts’ input, and timetables and cost estimates were adhered to. There was however a view that it was likely that ‘expensive’ expert’s fees could be increasingly capped, even in high value cases.

Discussion In literature, such as McIvor (2011)15 and Hyde (2011),16 there were suggestions that the motives for the reforms favoured the insurance industry and short-term government cost-cutting. This feeling was also strongly inferred by a number of claimant contributors. There was agreement among defendant contributors who favoured the reforms due to their anticipated positive impact on competition (through the costs-management and proportionality focus) and Bowman (2012)17 and Crouch (2011:17–19)18 where costs were said to have been too high, particularly on the claimant side. These contributors felt that the reforms would introduce a more robust approach to case management, as per Steward and Pratt (2013:23–5)19 and make law firms run more efficiently, as Regan (2013)20 had suggested. Most contributors felt that courts were likely to enforce restricting use of expert evidence. This fits with Lord Dyson’s suggestion that, post Jackson, courts may reach decisions based on less evidence than they have in the past (Solicitors Journal

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2013:3),21 although the research suggests this was not expected to be significantly so for higher-value cases. Regarding cost-budgeting requirements, on the whole contributors (both claimant and defendant) had concerns about tough sanctions for failure to comply with costmanagement rules and budgeting requirements, as outlined by Robins (2012:18– 21)22 and Kennedys (2013),23 as well as the associated professional dangers, highlighted by Cooper (2011).24 All contributors echoed concerns raised by Moore Stephens (2013)25 over the feasibility of expert witnesses providing costs’ estimates from the outset. Contributors’ views regarding apprehension of using SJEs agreed with Pugh and Pilgerstorfer (2005)26 and Solon (2012),27 which found that SJEs were only suitable for rare, unusually demanding and small cases. Also, contributors mentioned confusion over the implications of the reforms because judges did not seem to have received comprehensive training; this agreed with Hyde’s (2013)28 criticism of any such training judges had received on the reforms. Further, there was an observed lack of practice directions, as discussed by Solon (2012:596).29. Where contributors were optimistic, perceptions of the long-term impact of the reforms echoed Robins (2012:18–21),30 suggesting legal firms would find a way to secure their future, and Gibb (2013),31 expecting the personal injury industry to restructure itself to accommodate the changes. The findings were also in line with Master of the Rolls Lord Neuberger’s prediction that courts would be dealing with satellite litigation in the coming year and proportionality law would need to be developed on a case-by-case basis (Davidson 2012:6–11).32 Limitations There are some limitations to this study to be taken into account when considering the recommendations. First, this was a relatively small-scale study, involving only 25 firms. Additionally, due to access issues, the study was not fully representative of the views of all stakeholders – barristers/judiciary, insurance industry, interest groups, expert witness groups, etc. It was also noted by contributors that they were still very early into the Jackson reforms to be able to accurately determine much of the impact, based on experience. So the findings, in this respect, were expressed more as contributors’ feelings, opinions and related predictions, rather than on factual data based on actual experiences of the Jackson reforms and related events. Recommendations There appear to be opportunities for experts to demonstrate to lawyers that they can comply with the implications of the Jackson reforms, by becoming more pro-active about costs at the start of a case; and by stressing the value they may add, in order to assist with recoverability of fees, and by clearly showing efficiency and justification of their costs. It will be hard to change cost budgets once they have been submitted, so experts ought to increasingly present a realistic (and high enough for different eventualities) estimate up front. It is becoming increasingly important for experts to manage deadlines and review systems in place to alert of case deadlines where there are a number of cases running concurrently; also, to ensure that the instructing solicitor is adequately informed, on a timely basis, of any issues that may result in a report being served late, to allow the solicitor to act to avoid sanctions where possible. For medical experts, with a high volume of medico-legal work, it may be necessary to decrease the number of cases

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taken on, to fit with stricter timetabling, without necessarily being able to increase prices to compensate. Regarding case-dealing and adherence to stricter timetabling, given the new sanctions and professional risk for experts of not meeting court deadlines (Nugent 2013), it would be advisable for experts to update their ‘terms of reference’ with the client to ensure they are ‘Jackson-proof’ regarding liability (if, for example, an expert’s late report meant that such expert evidence was then struck out).

Conclusions The key finding of the study was that the reforms did appear to represent a changing landscape in the personal injury and clinical negligence market where expert evidence is used, but that there was opportunity for forward-thinking experts to adapt to meet the new order. The legal industry appeared to have been undergoing much structural change in recent years and perhaps Jackson had formalised this and will create the ‘disruption’ necessary to enable more powerful, positive changes. Such market disruption would point to opportunities for medical experts to take advantage of some of these changes and adapt their business models – concerning timing, offering and pricing – to fit with lawyers’ emerging future needs for medical experts. This is in order to demonstrate to lawyers that their preferred experts can comply with the implications of Jackson. For experts instructed on high-value, complex cases, there did not appear to be much anticipated change, as long as the use of such experts was fully justified and court timetabling adhered to.

Acknowledgements The research project was kindly sponsored by Monahans Forensic Services, Chartered Accountants who provide quantum accountancy expert services.

References Justice (2013). Justice.gov (online). Available at http://www.justice.gov.uk/civil-justice-reforms (last accessed 7 July 2013]. 2 Legal World News. Royal assent for Jackson Bill. New Law Journal 2012;162(7512):596. 3 Solicitors Journal. Jackson reforms to ‘re-establish equality of arms’. Solicitors Journal 2013;157 (13):3. 4 McIvor C. The impact of the Jackson reforms on access to justice in personal injury litigation. Civil Justice Quarterly 2011;30(4):411–28. 5 Crouch, A. (2011). ‘Shaping up: The picture taking shape in the Jackson reforms will create financial risks for barristers litigating personal injury claims and could be detrimental to client relationships’. Bar Focus. November, pp.17-19. 6 Steward G, Pratt J. There’s a new sheriff in town: the Jackson reforms and the new culture of ‘robust’ case management. The In-House Lawyer 2013;(212):23–5. 7 Ibid. n 4. 8 Lord Woolf (1996). Access to Justice: Final Report, the National Archives. Available at http://webarchive.nationalarchives.gov.uk/+/http://www.dca.gov.uk/civil/final/sec3c.htm#c13 (last accessed 1 October 2013). 9 Solon M (2011). New Law Journal (online). Available at http://www.newlawjournal.co.uk/nlj/content/end-expert-frolics-expert-evidence (last accessed 12 September 2013). 10 Expert Witness (2013). Your Expert Witness (online). Available at http://www.yourexpertwitness.co.uk/component/content/article/26-expert-witness-bitesize/856jackson-reforms-the-nitty-gritty-is-finally-published (last accessed 7 July 2013). 11 Ibid. n 3. 12 Kennedys (2013). Kennedy’s Law (online). Available at http://www.kennedyslaw.com/article/postjacksonanexpertsduty/ (last accessed 1 October 2013). 13 Solon M (2012). New Law Journal (online). Available at http://www.newlawjournal.co.uk/nlj/content/question-time-1 (last accessed 1 October 2013). 1

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14 Burn

S. Case management and evidence. Law Gazette 2013:21–2. n 4. 16 Hyde J. Judiciary ‘not ready’ for Jackson reforms. Law Society Gazette 2013. 17 Bowman C. Injured feelings: Jackson reforms. Law Society Gazette 2012. 18 Ibid. n 5. 19 Ibid. n 6. 20 Regan D (2013). New Law Journal (online). Available at http://www.newlawjournal.co.uk/nlj/content/new-normal (last accessed 1 March 2013). 21 Ibid. n 3. 22 Robins J. Shifting landscape: the Jackson reforms coming into force in eight months’ time. Cilex Journal 2012:18–21. 23 Ibid. n 12. 24 Cooper P. Jones v Kaney: will it prove to be a curse on some expert witnesses? The Expert Witness Institute Newsletter 2011:11–14. 25 Moore Stephens (2013). Moore Stephens (online). Available at http://www.moorestephens.co.uk/Controlling_expert_witness_costs.aspx (last accessed 7 July 2013). 26 Pugh C, Pilgerstorfer M (2005). Expert Evidence: The Requirement for Independence. London: Old Square Chambers. Available at http://www.oldsquare.co.uk/pdf_articles/3100173.pdf (last accessed 1 March 2013). 27 Ibid. n 13. 28 Ibid. n 16. 29 Ibid. n 13. 30 Ibid. n 22. 31 Gibb F. The Jackson reforms – what will they mean. The Times 2013. 32 Ibid. n 29. 15 Ibid.

Contacts/correspondence Harriet Formby Email: harriet.formby09@bathspa.ac.uk

Intellectual property & copyright statement I as the author of this article retain intellectual property right on the content of this article. I as the authors of this article assert and retain legal responsibility for this article. I fully absolve the editors and company of JoOPM of any legal responsibility from the publication of my paper on their website. Copyright 2013. This is an open-access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Psycho-Legal Assessment of Chronic Pain and Symptom Validity: A UK Perspective Vicki L Hall DclinPsy

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Abstract There is a range of non-organic factors, which predict outcomes and complicate recovery in chronic pain conditions. This article reviews these factors within the context of a psycho-legal assessment of pain-related disability. Part of this assessment includes evaluating whether the measured disability relates to the injury and determining the validity of the assessment findings. An important factor to consider is, therefore, malingering but the British medico-legal system considers decisions about malingering to be a matter for the court to decide (Halligan, Bass & Oakly, 2003). A framework for assessing malingered pain-related disability has been established within North America. The application of this framework is explored within the context of the British medico-legal system.

A framework for the assessment of chronic pain In their seminal paper on malingered pain-related disability (MPRD), Bianchini, Greve & Glynn (2005) provide a framework for a psychological assessment of chronic pain. They propose that this assessment should be multimodal and involve an evaluation of the breadth of symptoms that can occur co-morbid with a pain condition (i.e. it should include consideration of emotional, cognitive, behavioural and physical factors). It should also consider whether these factors can be attributable to the injury and it is, therefore, essential to consider whether a range of premorbid psychological and contextual factors are affecting recovery and causing psychological complications (Greve, Bianchini & Ord, 2012).

The importance of psychological assessments It is often psychological or non-organic findings that carry the greatest value in predicting pain-related disability (PRD; Block, Ohnmeiss, Guyer, Rashbaum et al., 2001; Lancourt & Kettelhut, 1992). There are limitations with using physical factors alone to predict outcomes in PRD as objective physical findings have been shown to relate to pain but not self-reported disability (Peters, Vlaeyen, Weber et al., 2005). Furthermore, cervical and lumbar disc abnormalities have been found in some asymptomatic patients (Boden, Davis, Dina, Patronas et al., 1990) and the presence of an abnormality does not predict subsequent low back pain seven years later (Borenstein, O’Mara, Boden, Lauerman et al., 2001).

Psychosocial factors There is a range of psychosocial factors, which can predict PRD (for example, catastrophic thinking, somatisisation and depression). The effect of these factors on outcome is detailed below.

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Catastrophic thinking refers to a tendency to fear pain and have a fear-induced understanding about its meaning (Proctor, Gatchel and Robinson, 2000). It has been shown to predict pain intensity, emotional distress and disability even after controlling for physical impairment (Sullivan, Lynch & Clark, 2005; Severjens, Vlaeyen, van den Hout & Weber, 2001). Somatisisation reflects the expression of psychological problems manifest in physical complaints and a tendency to complain of, or develop, physical symptoms and illness when under stress. This may be measured by the Minnesota multiphasic personality inventory (MMPI-II); in particular, the hysteria (measuring a tendency to be prone to developing somatic symptoms in response to life stressors) and hypochondriasis scales (measuring a preoccupation with the body and disease states; Butcher et al., 1989). These MMPI-II scales have been shown to be a greater predictor of outcome than imaging and neurological signs. Spengler et al. (1990) found that these scales contributed 26% of the variance in outcomes whereas imaging studies contributed 19% and neurological signs only 3%. Similarly Block et al. (2001) found that MMPI-II hysteria scale was the strongest predictor of clinical outcome exceeding medical factors (e.g. length of injury, type of surgery, number of previous spine surgeries). Although a newer version of the MMPI has been developed (MMPI-RF; Ben-Porath, Tellegen & Pearson, 2008), the research evidence base has not been substantiated to the same extent as the MMPI-II. Depression commonly occurs co-morbid with chronic pain presentations and has been found in between 18% to 56% of cases, with higher rates in patients seeking treatment (Von Korff, Crane, Lane et al., 2005; Miller & Cano, 2009; Dersh, Gatchel, Mayer et al., 2006; Mayer, Towns, Neblett, Theodore & Gatchel, 2008). It has been shown to be predictive of level of disability (Pincus et al., 2002; Linton, 2000); twiceas-long sick duration (Currie & Wang, 2004); a greater intensity, duration and frequency of pain complaints (Bair, Robinson, Katon & Kroenke, 2003); and it is predictive of return to work in lumbar discectomy patients, whereas clinical findings (e.g. MRI and physical examination) are not (Schade, Semmer, Main, Hora & Boos, 1999). Childhood adversity may refer to a range of adverse experiences (e.g. abuse, neglect, a chemically dependent caregiver or abandonment) that have occurred during childhood. Such experiences have been shown to be predictive of mental health problems (van der Vegt, van der Ende, Ferdinand & Verhulst, 2009) and somatisation (Spitzer, Barnow, Gau, Freyberger & Grabe, 2008; Imbierowicz & Egle, 2003). It also predicts pain symptoms (Davis, Luecken, & Zautra, 2005); unsuccessful surgical outcomes (Schofferman, Anderson, Hines, Smith & White, 1992) and has been shown to be related to early retirement in a survey of 8,817 working-age adults (Harkonmanki et al., 2007).

Contextual factors There is a range of contextual factors (e.g. occupational and educational factors, compensation and experience of treatment), which can cause psychological complications in chronic pain and affect outcomes. The type of job and the level of job satisfaction have been found to be related to outcomes. For example, physical workload, monotonous jobs and job satisfaction are all indicators of back pain, work absences and disability (Bigos et al., 1991; Hagen et al., 2002; Bongers et al., 1993). Job satisfaction has also been shown to predispose a person to develop pain problems in asymptomatic patients (Bigos et al., 1991), although some caution needs to be taken when using this factor to predict PRD as

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other studies have shown it does not predict outcomes (Hartvingsen, Lings, Leboeuf & Bakketeig, 2004). A lower level of education has also been found to be related to a longer duration and higher reoccurrence of back pain (Dionne et al., 2001). A person’s experience of treatment can also affect chronic pain outcomes. For example, low satisfaction with care (Wickizer, Franklin, Fulton-Kehoe, Mootz & Smith-Weller, 2004) and delays in treatment (Crook & Moldofsky, 1994) have been shown to result in worse outcomes. When a person is involved in a compensation claim, they may have delays in treatment for a number of reasons and being involved in a compensation claim has also been show to relate to significantly higher rates of pain, depression and disability (Chibnall & Tait, 1994).

Malingering One reason for compensation to result in worse outcomes is that the person may deliberately feign or exaggerate symptoms for financial gain. This is referred to malingering, which is the intentional exaggeration or feigning of symptoms for an external reward (e.g. medication, avoiding work or financial gain) (APA, 2013). The prevalence of malingering in patients with pain is 20–50% (Mittenberg, Patton, Canyock & Condit, 2002; Greve, Ord, Bianchini & Curtis, 2009; Kay & Morris-Jones, 1998). Malingering is, perhaps, the most problematic outcome to assess, particularly within the British medico-legal system and, therefore, requires further elaboration.

The psychological and financial costs of malingering Within the British medico-legal system, clinicians will often not diagnose malingering because making decisions about malingering is seen as a matter which should be left for the court to decide (Halligan, Bass, & Oakley, 2003). Malingering, therefore, often goes undiagnosed but there are potential costs to the patient that are associated with this – there is evidence that malingerers may engage in potentially self-injurious behaviour; for example, there are incidences of patients diagnosed with malingered pain-related disability who have undertaken surgery (Bianchini, Heinly & Greve, 2004; Bianchini & Greve, 2009). There is evidence that patients who are malingering will come to take on these fabricated symptoms after they have stopped malingering as they will unconsciously internalise the symptoms (Merten & Merkelbach, 2013) and we know that if a person is treated as though they have symptoms related to an injury when they do not, they can develop an iatrogenic reaction and unconsciously display these symptoms (Suhr & Gunstad, 2002, 2005). There are also financial costs with not detecting malingering. An American study estimated that fraudulent mental disorder social security disability claims cost $20.02 billion. These costs were based on mental disorder data published by the Social Security Administration (Chafetz & Underhill, 2013). There is no equivalent data for the UK but the UK Department of Work and Pensions 2012 statistics show that there were 3.28 million recipients of Disability Living Allowance (DLA) and £1.2 billion, of total benefit expenditure is overpaid due to fraud (Department of Work & Pensions, 2013). This is likely to be only the tip of the iceberg as the validity of claims is not routinely screened.

There are also, of course, potentially high costs to be paid in terms of any false diagnosis of this condition as it is a criminal offence if it is related to fraud and it is also considered a pejorative label. Malingering or symptom exaggeration can also be the consequence of the pressure of the litigation process. This process may make

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claimants feel overwhelmed as they may be faced with having to convince others that their problems are real because it appears as though others do not believe their problems.

Assessing Malingering A framework has been devised for detecting MPRD (Bianchini et al, 2005). The diagnostic criteria for probable MPRD includes the following: the person must have evidence of a significant external incentive (criterion A) and meet two or more types of evidence from physical evaluation (criterion B), cognitive and perceptual testing (criterion C), and evidence from self-report (criterion D). The two or more types of evidence from criteria B–D must come from well-validated measures with error rates. To meet the criteria for MRPD, it is also necessary to fulfil criterion E, which specifies that the behaviour meeting necessary criteria (B–D) is not fully accounted for by psychiatric, neurological or developmental factors. Pivotal to these criteria is the application of symptom validity tests (SVTs), as these tests provide evidence, which is well validated with error rates.

Symptom validity tests SVT is a superordinate term for all methods that tap negative response bias in both symptom production (including underperformance in neuropsychological tests) and symptom over-reporting (Greve, Bianchini and Brewer, 2013). PVTs are cognitive tests designed to detect ‘performance invalidity’ on neuropsychological test batteries. These are designed to be insensitive to severe cognitive impairment and sensitive to ‘malingering’. 29% of litigating chronic pain patients have been shown to fail these measures (Meyers & Diep, 2000). They are pivotal to the MPRD criteria as they are the best-calibrated and most well-validated SVTs. Self-report SVTs (criterion D) are designed to be insensitive to symptoms reported by bona fide patients and sensitive to malingering. They are validated using clinical patients who have been diagnosed to be malingering, using explicit criteria (e.g. Bianchini et al., 2005, MPRD) or groups of individuals who are asked to feign symptoms. These groups are then compared to etiologically similar patients with bona fide symptoms. The Bianchini et al. (2008) criteria for MPRD specifies that ‘intent’ is defined by the combined improbability of a person failing more than one of these measures and belonging to a group of chronic pain patients with bona fide symptoms. Failure on one SVT failure is not uncommon in non-malingering control group samples when multiple SVTs are administered (Larrabee, 2003; Meyers & Volbrecht, 2003; Victor, Boone, Serpa, Buehler & Ziegle, 2009). The threshold for diagnosing malingering is therefore set at failing more than one SVT in order to avoid false positive errors.

The application of SVTs in the UK In the UK medico-legal arena, experts may consider there to be a problem with directly applying SVTs within a ‘malingering’ framework (e.g. MPRD, Bianchini et al., 2005) as malingering is considered as a matter for the court to decide. Experts may still be asked to give an opinion about malingering and some UK experts will use malingering diagnostic frameworks to provide such an opinion (Bali v Farooq, 2012). However, many UK experts still avoid using this term as they consider it not to be a medical diagnosis, viewing the unmasking of malingering to be purely a legal process (Halligan et al., 2003). This may lead such experts to explain away SVT failures with non-malingering explanations, with little empirical or clinical support. For example, PVTs are sometimes called effort tests and, therefore, clinicians sometimes erroneously believe that they are sensitive to effort (McMillan, Anderson, Baker, Berger & Powell, 2009) and a PVT failure may be due to poor effort caused by depression or fatigue. However, there is no evidence to support the hypothesis that

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PVTs are sensitive to poor effort as they are designed so that persons with significant neurologic dysfunction can pass them, and therefore require very minimal levels of cognitive capacity and very little ‘effort’ (Larrabee, 2012; Heilbronner et al., 2009). Furthermore, PVTs are insensitive to conditions, which affect motivation, such as severe anxiety and depression (Ashendorf, Constantinou & McCaffrey, 2004; Iverson, LePage, Koehler, Shojania & Badi, 2007; Rees, Tombaugh & Boulay, 2001; Yanez, Fremouw, Tennant, Strunk & Coker, 2006). Studies have also found that pain, emotional distress and fatigue could not account for PVT failure (Busichio, Tiersky, DeLuca & Natelson, 2004; Etherton, Bianchini, Greve & Ciota, 2005). However, it may be possible that a person may not able to cooperate with the test procedures in some cases of very severe depression or severe executive difficulties. An individual may also fail an SVT due to unconscious processes causing them to over-report symptoms or affecting their cooperation with testing. However, it should only be possible to provide this as an explanation if there is evidence to support it (e.g. symptom over-report may occur in the context of somatisation disorders but this can only be inferred if an assessment of somatisation has been undertaken). Even if there is evidence for somatisation, clinicians should be mindful that PVT failure only occurs in a minority of these cases (Kemp et al., 2008) and there is no reason to assume that a somatisisation patient could not cooperate with testing on PVTs or have cognitive impairment that exceeded a severe head injury (Merten & Merkelback, 2013). It is also possible that a ‘cry for help’ may explain an SVT failure but clinicians should be particularly careful not to use such pseudo-psychological explanations without significant evidence to support them (Merten & Merkelbach, 2013). There is no reason to assume that this is a common cause of SVT failure because SVT failure is uncommon in a range of bona fide clinical groups who may be expected to ‘cry for help’ (e.g. traumatic brain injury, chronic pain, depression – see Larrabee, 2008, for review). As Iverson (2006) notes: ‘it could be considered biased if a clinician has a much lower threshold, and relies on much less evidence, to attribute exaggeration to a cry for help versus deliberate misrepresentation of symptoms and problems to influence the results of a forensic evaluation’. Furthermore, as clinicians we should be mindful that clinical evidence is required to attribute pseudo-psychological explanations to the tests because if we attribute these reasons to SVT failure we are going beyond the psychometric scope of the test. SVTs were developed to answer a specific psychometric question: what is the statistical likelihood that the person’s test result has come from a ‘malingering group’ as opposed to a range of bona fide clinical groups (e.g. pain, traumatic brain injury)? Although SVTs were intended to measure ‘malingering’, an SVT failure may, nevertheless, sometimes be attributable to other factors (e.g. factitious disorder, somatisisation). However, factitious disorder or somatisation does not rule out MPRD as to meet this criteria the SVT failure should not be fully accounted for by psychiatric, neurological or developmental factors. In fact, cases of pure malingering may be rare as symptom exaggeration may be underpinned by a range of external and internal factors, and there may be a range of conscious and unconscious factors affecting their presentation. Malingering may not, therefore, be considered to be binary phenomena but it occurs on a continuum: a continuum between selfdeception and other-deception; between external and internal gains (Merten & Merkelbach, 2013); and between no exaggeration and extreme exaggeration with malingering occurring at the end of a spectrum of exaggeration. Given the complexity of the diagnosis of malingering, it would be difficult for a court to make a decision about this without the input of an expert. However, in order to comply with court expectations, malingering may be considered as a differential

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diagnosis of exclusion but left as a decision for the court to decide. This exclusion criterion is consistent with the MPRD criteria, which states that the symptom exaggeration behaviours should not be fully accountable by psychiatric, neurological or developmental factors.

Conclusions There is a range of psychological factors, which predict pain-related disability. A thorough psychological assessment of chronic pain should be multimodal and involve an evaluation of emotional, cognitive, behavioural and physical factors; a consideration of whether these can be attributable to the injury (Bianchini et al., 2005) as well as an evaluation of contextual and psychosocial factors, which can cause psychological complications. In order to provide such an assessment, diagnoses such as malingering may need to be considered and there are empirical frameworks for assessing this using a range of SVTs. There are, however, concerns among experts about making decisions about ‘malingering’ in the UK medico-legal arena, as this is considered to be a decision for the court. Experts should be cautious about dealing with this dilemma by explaining away SVT failures with non-malingering causes without evidence to support their hypotheses. The interpretation of SVTs needs to be considered within the context of a full psychological assessment.

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Should the Law decide on moral issues? Lord Walker, Supreme Court Judge

British judges have often said that it is not the business of the court to adjudicate on the seriousness, cogency or coherence of theological beliefs or liturgical practices1. It has also often been said that the court is not a court of morals. "Moral issues" is the expression used in the title to this talk, and I do not suppose that any of us would regard morality as exclusively the concern of religious believers. But it is mostly in the context of religious beliefs and practices that the court is asked, increasingly often, to adjudicate on questions of conscience. The ever- growing volume of cases has two main causes. One is the Human Rights Act 1998 and other statutes aimed at unfair discrimination, including most recently the Equality Act 2011, which has consolidated and expanded earlier legislation. The other is our changing society, which is both more multicultural and (at least for those who would in the past have ticked the box for "C of E") less religious. British judges had no say in the enactment of the Human Rights Act. Parliament enacted it, though now both of the largest political parties seem to be having second thoughts about it. The judges must do their best to interpret it and apply it: that is what judges are for. This is a huge and, for me, fascinating subject. I could talk about it for hours. Fear not, it has been a long day and I am not going to speak for long. I propose to look at a few key issues rather than attempting the impossible task of covering the whole field. Article 9 of the European Convention on Human Rights is concerned with freedom of thought, conscience and religion. Under the Human Rights Act public authorities must act consistently with Convention rights. Article 9 draws a very important distinction between freedom of belief as such (which is unqualified) and freedom to manifest one’s “religion or belief, in worship, teaching, practice and observance”. This right is qualified by Article 9, paragraph 2: “Freedom to manifest one’s religion or belief shall be subject only to such limitations as are prescribed by law and are necessary in a democratic society in the interests of public safety, for the protection of public order health or morals, or for the protection of the rights and freedoms of others.” Manifestation of religion can take many different forms. Some are concerned with very personal matters: circumcision, hairstyle, diet, dress. It is easy to recognise a man who is a Sikh, or an Orthodox Jew. Other manifestations of belief are communal. Almost all religious communities worship in public, and their acts of worship are often accompanied by music and singing, and sometimes by bells, incense, vestments and processions. These personal and communal observances do sometimes lead to friction and difficulties with authorities, with employers, with neighbours and even within families. 1

R(Amicus) v Secretary of State for Trade and Industry [2004] IRLR 430, 438 Journal of Observational Pain Medicine – Volume 1, Number 3 (2014) ISSN 2047-0800


Issues of this sort most often arise in schools, in employment and in decisions about the care and upbringing of children. But I would like to start with a case decided by the Supreme Court of Canada in 20042. The Canadian Charter of Rights contains provisions similar to those of Article 9 of the European Convention, although expressed in different terms. The Canadian case was in form a dispute between landlord and tenants, the tenants being several Orthodox Jews with apartments in a luxury condominium in Montreal. Each of them wished, in breach of the condominium regulations, to erect a succah on his balcony or patio during the Festival of Succot. A succah is a small temporary hut and Succot is a nine-day festival celebrating the Jewish people’s forty years of wandering in the desert. During the festival an Orthodox Jew is required to treat the succah as his principal abode. The Supreme Court of Canada split 5-4 over the interpretation of the expert evidence, and in particular whether religious law required the succah to be on the participant’s own property, or whether a communal succah (available outside the condominium) was sufficient. Here Canada’s highest court found itself drawn into just the sort of investigation which secular courts are not equipped to undertake. Yet in the balancing exercise required by any decision on qualified rights, it seems inevitable that the court should consider whether a particular religious observance is of central importance to that faith, or is more in the nature of an optional extra. In the United States Supreme Court3 Justice Scalia put the point vividly: “Dispensing with a ‘centrality’ inquiry is utterly unworkable. It would require, for example, the same degree of ‘compelling state interest’ to impede the practice of throwing rice at church weddings as to impede the practice of getting married in church.” That was in a case about the legality of the use of a banned substance for religious purposes by members of the Native American Church. Clothing and ornament often raise this issue. The religious importance to a male Sikh of wearing a turban is well recognised; in the United Kingdom Sikh men are exempt from the statutory obligation to wear a crash helmet when riding a motorbike.4 But for a Christian wearing a cross or crucifix is not, so far as I know, a matter of obligation for any denomination. Indeed some denominations would regard it as quite unsuitable. And many young people who wear a cross as a fashion accessory have little idea of its reference to the events of Good Friday. The English Court of Appeal5 recently rejected a claim for indirect discrimination brought by an airline check-in desk worker who was prevented by her employer’s uniform dress policy (who prohibited any ornament worn on the neck) from wearing a silver cross. The court’s attitude to employees who object on religious grounds to the employer’s dress code, or to an employer’s refusal to let them have time off for religious observances, may appear a bit ungenerous. For an employer to react in terms of “if you do not like it get another job” is not in line with best industrial practice. Against that it may be said that many people with strong religious convictions tend to be (in a literal sense) uncompromising, since they may see compromise as a betrayal of their faith.

2

th

Syndicat Northwest v Anselem (2004) 241 DLR (4 ) 1 Employment Division of Oregon v Smith 494 US 872, 888 4 Road Traffic Act 1988 Section 16(2); but not in Canada: R v Badesha 2008 ONCJ 94 5 Eweida v British Airways plc [2010] EWCA Civ. 80; [2009] 1RC 927 3

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At present the most important area of controversy is probably the muslim veil. It may be a problem in schools, in the workplace, in hospitals or in court. The fullest discussion is in a case decided by the House of Lords in 20066. Reversing the Court of Appeal, they upheld the policy on school uniform decided on by a head teacher and the governors who were predominantly Muslim. The complainant was at the time a fourteen-year old Muslim schoolgirl with a nineteen-year old brother who was (since her father’s death) the head of the family. The school had a very diverse intake, with twenty-one different ethnic groups and ten religious groupings (Muslims being predominant). The head teacher and governors had carefully reviewed and consulted on the school policy on uniform dress, which permitted three options for girls. All this is set out in detail in Lord Bingham’s opinion.7 Having accepted the policy for two years the complainant then came to school, accompanied by her elder brother, wearing a long coat-like garment called a jilbab. There was a standoff and litigation ensued. All the speeches in the House of Lords are of interest, but I particularly commend that of Lady Hale, who is at present (unfortunately) the only woman in the Supreme Court. She pointed out that for a woman to wear muslim dress in western society may be a sign of autonomy, not subservience. Having developed this point she continued8: “But schools are different. Their task is to educate the young from the many and diverse families and communities in this country in accordance with the national curriculum. Their task is to help all of their pupils achieve their full potential. This includes growing up to play whatever part they choose in the society in which they are living. The school’s task is also to promote the ability of people of diverse races, religions and cultures to live together in harmony. Fostering a sense of community and cohesion within the school is an important part of that. A uniform dress code can play its role in smoothing over ethnic, religious and social divisions.” In general parents are entitled to bring up children in accordance with their own religious beliefs. But they have a higher duty, in the eyes of the law, to safeguard their children’s health and welfare. The classic dilemma, found in cases all over the world, is of parents who are Jehovah’s Witnesses and refuse to permit a child to have a blood transfusion, even when his life is in imminent danger. Such cases have occurred in New Zealand,9 Canada,10 England11 and elsewhere. In all these cases the court has intervened to overrule the parents’ refusal to agree to life-saving treatment. The need for a life-saving blood transfusion is the most dramatic instance of the court having to balance a parent’s religious convictions against a child’s welfare. Comparable decisions have to be made from time to time, although not with life at stake, when parents disagree on other matters, such as whether their son should be circumcised for religious (rather than medical) reasons. This problem was fully considered by the English Court of Appeal in 2000.12 The boy (then aged five) had a Turkish “not very observant” Muslim father and an English non-observant Christian mother. They had met when she was on holiday in Turkey. The marriage lasted less

6

R(Begum) v Denbigh High School Governors [2007] 1AC 100 Paras 3 to 8 8 Para 97 9 Re J (An Infant) [1996] 2 NZ LR 134 10 th B (R) v Children’s Aid Society of Metropolitan Toronto (1995) 122 ELR (4 ) 1 11 Re S [1993] 1 FLR 376 12 Re J (Specific Issue Orders: Child’s Religious Upbringing and Circumcision) [2000] 1 FLR 571 7

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than four years and the mother brought up their son in a secular English environment. The Court of Appeal upheld the judge’s order that the boy should not be circumcised. The judge made that order for four reasons: the boy was having an essentially secular upbringing in England; the procedure was irreversible, had no medical basis, and involved a “small but definable” medical and psychological risk; the boy was vulnerable because of the hostility between his parents; and the mother, the primary carer, was strongly opposed to it. In short, the Court respects a parent’s religious views, but treats the child’s welfare as paramount, and does not unrealistically attribute the same religious views to a young child. The situation becomes more difficult if the child is of an age when religious beliefs have begun to form. Another division of the English Court of Appeal had a poignant case13 about a nine-year old boy whose father (then a widower) had been “withdrawn from” (that is ostracised) by the Exclusive Brethren within whose community the boy had been brought up for more than half his life. The community had extremely strict rules about keeping themselves to themselves, and on the father’s expulsion the child began to be looked after by other members of the community, who applied for a residence order. The court welfare officer who interviewed the boy said that he was close to his father; but at the same time he felt himself to be a part of that community, and wished to continue to be part of it. The judge did not accept that the boy had been “brainwashed” but did observe, “He has been taught their beliefs and their separatist doctrine virtually every day of his life since [he was four].” The judge’s view was that he had to choose between the brethren and the father; there was no middle course. And despite the child’s own wishes the judge chose the father, and the Court of Appeal upheld that decision. It is perhaps fair to say that on matters of religion the court seems to incline to a policy of “pas trop de zele.” The more extreme the religious opinions and observances to which a growing child may be exposed, the more wary the court becomes. For those of you with a taste for legal and social history it is interesting to compare the last decision with a decision of the Court of Appeal in Chancery, in exercise of its ancient wardship jurisdiction, about 150 years ago. In Stourton v Stourton14 the ward was a ten-year old boy of delicate constitution, the posthumous son of the Honourable John Stourton, the younger brother of Lord Stourton. They were a prominent Roman Catholic family and the child’s mother had been a Catholic, but in 1852 (when her son was five) she became a member of the Church of England and brought up her son in that faith. Four years later (a delay of which the court was very critical) Lord Stourton started wardship proceedings, asking that his nephew should be brought up as a Roman Catholic. The Master of the Rolls dismissed that application, confirming the widow as the boy’s guardian, and the Court of Appeal in Chancery upheld that order. The case is particularly interesting, a century and a half later, in terms of what has changed and what remains the same. What has changed is the absence of any sort of confidentiality in the reporting of family proceedings, the court’s readiness (even at

13 14

Re R(A minor) (Residence: religion) [1993] 2 FLR 163 (1857) 8 De GM & G 760

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appellate level) to take evidence from a vulnerable witness, and the biblical prose in which the interview was described:15 “He [this is, remember, a ten-year old boy] spoke on the subject of transubstantiation, the attributes of the Virgin Mary, the invocation of saints, and the authority of the Pope, in a manner convincing me that the Protestant seed sown in his mind has taken such hold, that if we are to suppose it to contain tares, they cannot be gathered up without great danger of rooting up also the wheat with them.” The Court seems to have struggled between its respect for an ancient peerage (“a name of such distinction as rank and antiquity can give”) and its barely-suppressed horror that an English peer could think of “placing his brother’s son at a Jesuits’ seminary or establishment.” But what has not changed is the essential good sense of the Court’s general approach:16 “The first and paramount duty of the Court unquestionably is to consult the well-being of the infant, and in discharging that duty the Court recognises no religious distinctions.” So these problems are far older that the Human Rights Act, but it has certainly increased the volume of litigation in this area.

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15 16

Knight Bruce LJ at pp767-768 At p771

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