Review Article Phantom Limb Pain and Its Psychologic Management: A Critical Review ---

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Shruti Niraj, MSc,* and G. Niraj, MD, FFPMRCA†

ABSTRACT:

Phantom limb pain is a puzzling phenomenon, from the viewpoints of both the patient experiencing it and the clinician trying to treat it. This review focuses on psychologic aspects in the origin of the PLP and critically evaluates the various psychologic interventions in the management of PLP. Whereas pharmacologic and surgical treatments often fail, psychologic interventions may hold promise in managing PLP. Studies using cognitive-behavioral therapies and hypnotherapy are reviewed. The outcome reports for psychologic therapies have been mainly positive. The results of the majority of these studies show a reduction in PLP. However, the lack of well controlled and randomized trials makes it difficult to draw firm conclusions regarding the effectiveness of these psychologic therapies in the treatment of PLP. Ó 2014 by the American Society for Pain Management Nursing

From the *Leicestershire Partnership Trust; †Leicester General Hospital, Leicester, United Kingdom. Address correspondence to Shruti Niraj, MSc, Psychological Wellbeing Practitioner, Leicestershire Partnership Trust, Towers Hospital, Gipsy Lane, Leicester LE5 0TD, UK. E-mail: [email protected] Received January 8, 2008; Revised April 1, 2012; Accepted April 3, 2012. 1524-9042/$36.00 Ó 2014 by the American Society for Pain Management Nursing doi:10.1016/j.pmn.2012.04.001

The phenomenon of phantom limb has always fascinated surgeons, psychiatrists, and psychologists and has been studied extensively. Although this condition is easy to diagnose, the pathophysiology remains unclear and it can be difficult to treat successfully. The amputation of a limb is commonly followed by a sensation that the amputated part is still present. The perception of the lost limb is so real that not until the amputees lift the bed sheets to see it do they realize it has been cut off (Katz, 1993). Weir Mitchell coined the term ‘‘phantom-limb’’ in 1872 to describe the persisting sensory awareness of a limb after amputation (Louis & York, 2006). Phantom limb refers to the sensations in a part of the body that has been deafferented or amputated (Flor, 2002). Virtually all amputees experience phantom sensations, which rarely pose a clinical problem. However, some amputees experience severe pain in the body part that is no longer present, which may be extremely difficult to treat. Phantom limb pain (PLP) is a type of neuropathic pain that is perceived in the territory of the amputated limb. Although such pain is most common in patients after limb amputation, it can also occur after the surgical removal of other body parts, such as breast, rectum, penis, testicles, eye, tongue, or teeth (Flor, 2002). PLP is a condition that has baffled health professionals, and its management has been ineffective at the best. An understanding of the underlying mechanisms of PLP is likely to lead to novel and rational types of treatments. Pain Management Nursing, Vol 15, No 1 (March), 2014: pp 349-364

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The aim of the present review was twofold: 1) to critically examine the role of psychologic factors in the origin of PLP; and 2) to review the current literature on the treatment of PLP by psychologic therapies.

PATHOPHYSIOLOGY OF PHANTOM LIMB PAIN A number of theories have been offered to explain the origin of phantom limb phenomena. Although the exact mechanism for the development of PLP has not been clearly elucidated, there are three theoretical pathways proposed. They are the psychogenic, peripheral, and central neural mechanisms. Psychologic Factors The behavioral science literature usually characterizes the PLP as being a manifestation of either some mentalemotional problem or as one of the basic personality structure. Parkes (1973) found that amputees with persistent phantom pain scored highly on a personality measure of ‘‘compulsive self-reliance’’ and ‘‘rigidity.’’ This theory has not gained widespread acceptance, although a few researchers have uncritically acknowledged it despite the presence of any empirical evidence supporting it (Dawson & Arnold, 1981; Dernham, 1986). More recent research suggests that psychologic factors do not play a causal role in PLP (Arena, Sherman, Bruno, & Smith, 1990). Rather, it is becoming increasingly clear that under certain circumstances psychologic factors such as depression, anxiety, fatigue, and insomnia may trigger and exacerbate the PLP in psychologically healthy individuals (Arena, Sherman, Bruno, & Smith, 1990; Giummarra & Moseley, 2011; Sherman, Sherman, & Gall, 1987). Loss of a limb for whatever reason is a major event with profound implications for the psychologic health of the individual involved. That is why it is not surprising that 20%-60% of amputees are assessed as being clinically depressed (Shukla, Sahu, Tripathi, & Gupta, 1982; Whyte & Niven, 2001). Another study has reported that amputees with depressive symptoms were more likely to characterize their pain as more severe than those without depressive symptoms (Ephraim, Wegener, Mackenzie, Dillingham, & Pezzin, 2005). Cognitive factors also play a part in the modulation of PLP. Patients who lack coping strategies and fear the worst when confronted with episodes of pain are more affected by the pain and report more interference than patients who cope well with the situation (Hill, Niven, & Knussen, 1995). Psychologic variables before the amputation are also predictive of

PLP. Patients who received less support before the amputation tend to report more PLP (Gallagher, Allen, & MacLachlan, 2001). Peripheral Factors Recent evidence suggests that chronic phantom pain is usually a series of complex type of referred pain with very real physiologic bases. Theories focusing on the peripheral nervous system suggest that phantom pain occurs due to the abnormal discharges at dorsal horn of the spinal cord caused by a loss of afferent input from the amputated limb (Flor, Nikolajsen, & Jensen, 2006). After an amputation, when peripheral nerves are cut or injured, fibers from the cut end of the nerves grow into nodules or neuromas. Such neuromas generate spontaneous and abnormal activity, which is thought to be a potential source of the stump pain and PLP (Flor, Nikolajsen, & Jensen, 2006). This theory is further supported by studies reporting a reduction in phantom pain with drugs blocking sodium channels (Borghi, d’’Addabbo, White, Gallerani, Toccaceli, Raffaeli, . Mercuri, 2010; Karanikolas, Aretha, Tsolakis, Monantera, Kiekkas, Papadoulas, . Filos, 2011). However, this theory fails to explain the mechanism of PLP in patients with congenital absence of limbs (Flor, Nikolajsen, & Jensen, 2006; Weeks, Anderson-Barnes, & Tsao, 2010). Central Neural Mechanisms There are three central neural mechanisms that have been elucidated. They are cortical reorganization theory, spinal cord sensitization theory, and neuromatrix theory. Cortical reorganization is the most accepted and widely cited mechanism for PLP. During reorganization, the cortical areas representing the amputated limb are taken over by the neighboring representational zones in both the somatosensory and motor cortex (Baron, Binder, & Wasner, 2010; Flor, Nikolajsen, & Jensen, 2006; Ramachandran, Brang, & McGeogh, 2009). Cortical reorganization partly explains why the afferent nociceptive stimulation of neurons within the stump produces sensation in the missing limb. The extent of cortical reorganization has been found to be directly related to the degree of pain and the size of the deafferentiated region. This has been correlated by many imaging studies (Flor, Nikolajsen, & Jensen, 2006; Roux, Ibarrola, Lazorthes, & Berry, 2008; Spring, 2010). Spinal cord sensitization occurs after injury to the peripheral nerve. There is increased neuronal activity, expansion of the neuronal receptive field, and hyperexcitability of the spinal neurons. This process is called central sensitization. This is followed by up-regulation

Phantom Limb Pain

of the spinal cord receptors and is termed ‘‘windup phenomenon’’ (Baron, Binder, & Wasner, 2010). There is concomitant reduction in descending inhibitory transmission from the supraspinal centers. The combination of these factors has been proposed to result in the generation of PLP (Baron, Binder, & Wasner, 2010; Costigan, Scholz, & Woolf, 2009). Melzack (2005) proposed that within the brain lies a ‘‘neuromatrix,’’ i.e., a network of neurons in several brain areas, including the thalamus, somatosensory cortex, the reticular formation, the limbic system, and the posterior parietal cortex, that responds to sensory input and constantly produces a specific pattern of impulses, called the ‘‘neurosignature,’’ which registers that the body is intact (Melzack, 2005). The deprivation of various inputs from the limbs to the neuromatrix causes an abnormal neurosignature to be produced that results in the generation of PLP (Iannetti & Mouraux, 2010; Melzack, 2005). It does not explain why some individuals with amputations develop PLP and others remain free of pain. Nevertheless, the brain areas suggested in the neuromatrix theory are significantly involved in the experience of phantom limbs and PLP. Summary of Pathophysiologic Factors The above review of underlying factors indicates that a number of mechanisms are involved in generating the PLP and that these include changes in the peripheral nervous system, spinal cord and brain. The initiation of phenomenon takes place owing to the changes arising in the peripheral nervous system, which subsequently generates a flow of events that sweep more centrally, leading to reorganization of cortical brain structures. These changes may be responsible for the complex and vivid sensation that characterizes certain phantom pain sensations. Psychologic factors do not seem to contribute to the causation but may instead affect the course and the severity of the pain.

TREATMENT OF PHANTOM LIMB PAIN There is no single best treatment for PLP. A survey of literature by Sherman, Sherman, and Gall (1980) identified 68 different methods of treating PLP, 50 of which are still in use. However, several studies have shown that most treatments for PLP are ineffective or appear to be transiently effective; follow-up usually reveals long-term benefits to be minimal, with levels of relief below those expected with placebo treatment alone (Sherman, Sherman, & Gall, 1980). There is a lack of evidence to support the efficacy of many treatments, because most of the studies suffer from major methodologic errors, such as small sample size, insufficient

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randomization, and short follow-up periods. Taken together, the results of these studies suggest that PLP is one of the most undertreated chronic pain syndromes. Psychologic Treatment Although psychologic factors do not seem to be causative factors in PLP, the benefit of psychologic treatments may be to reduce severity. Despite considerable efforts, medical and surgical treatments often fail to relieve PLP. Patients with chronic pain usually become ‘‘problems’’ in the health care system, and psychologic consultation may be requested (Linton, 1982). Given that cognitive and affective processes may trigger or exacerbate PLP, it is important that patients be prepared before amputation for the presence of phantom limb. Patients who are ill prepared for amputation suffer needlessly with PLP and concern about their sanity (Katz, 1993). Patient education programs and treatment of stress before the amputation have become the standard practice. The common psychologic therapies in the management of PLP are cognitive-behavioral therapy and hypnotherapy. The present review focuses on the studies involving limb amputation and brachial plexus avulsion, because these are the common causes of PLP. Hypnosis Hypnosis has been used as a psychologic technique for treatment of a broad range of disorders and illnesses, such as depression, phobias, psychotic disorders, posttraumatic stress disorders, and somatization disorders (Montgomery, DuHamel, & Redd, 2000). The range of painful conditions whose management has been facilitated with the use of hypnotic technique is incredibly broad, ranging from iatrogenic pain to intractable pain associated with chronic diseases (Chaves, 1986). Given the breadth of treatment contexts, hypnosis is best known as a pain management technique. Hypnosis usually includes visual imagery but implies a state of highly focused attention during which one is susceptible to direct or indirect suggestions for reaching a goal. For the purpose of pain relief, suggestion is a central component of clinical efficacy (Turk & Gatchel, 2002). Hypnotic procedures using suggestion and imagery have a long and established history of alleviating a range of painful conditions (Hilgard & Hilgard, 1975). Two meta-analyses on the efficacy of hypnosis in pain management have concluded that hypnotic suggestion can act as an effective analgesic (Hawkins, 2001; Montgomery, DuHamel, & Redd, 2000). Available evidence for the efficacy of hypnosis for the treatment of pain was considered as level I evidence in the light of the National Health and Medical Research Council

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TABLE 1. Summary of Studies Using Hypnosis in the Management of Phantom Limb Pain (PLP) Study

Hypnotizability Assessed No

Chaves (1986)

No

Chaves (1993)

No

Brown et al. (1996)

No

Oakley et al. (2002)

No

Chan (2006)

No

Muraoka et al. (1996)

No

Treatment

Left above-knee amputation (pain before)— PLP for several weeks Amputation of arm (pain before)— PLP for 5 months felt as ‘‘tension’’ and continuous movements in ‘‘phantom hand and fingers’’ Mid-thigh amputation of right leg (pain before)— PLP for 4 years felt as ‘‘biting ants,’’ tight bands, muscle tension (phantom ‘‘leg’’ in uncomfortable position) Amputation of right leg at the knee (pain statue before not reported)— ‘‘severe’’ PLP (details not reported) Above-knee amputation of right leg (no pain before)— PLP for 6 years, began 2 years after the surgery, pain felt as:  ‘‘pins and needles’’ in phantom foot  ‘‘toes’’ in a vice  cutting pain in ‘‘foot’’  chiseling pain in ‘‘ankle’’ Below-knee amputation of left leg after a fall (no pain before)— PLP for 7 years, began 2 years after the amputation, pain felt as:  ‘‘pins and needles’’ throughout the ‘‘leg’’  ‘‘saw’’ cutting into left ‘‘toes’’  drill pushing into his left ‘‘heel’’

10 sessions (7 of hypnosis): relaxation, self-hypnosis, transfer of hypnotic (‘‘cold’’ imagery) glove anesthesia 3 hypnosis sessions: relaxation, tension reduction suggestions; warmth imagery; home use of hypnosis audiotape

Pain medication reduced to 50%; improvement in mood and activities Free of phantom pain and stump pain over 5-year follow-up; using tape once a month

Hypnosis and relaxation sessions (number not specified); suggestions of phantom shrinking; hypnotic images: ‘‘decapitate ants,’’ ‘‘cut bands’’; daily use of hypnosis audiotape

At the end of treatment, discomfort reduced to 30% of previous level; occasionally pain-free; phantom limb reported to be shrinking

Three session (5 hours in total) and an audiotape: hypnotic metaphor or tree damaged by flood water, losing branches, then regrowing stronger 1 hour (25 minutes of hypnosis) over 8 weeks: visualization of ‘‘special place’’; hypnotic imagery: ‘‘chiseller on holiday’’; ‘‘sea water loosening vice’’; positive images of change and progress

At 1-year follow-up, wearing prosthesis and engaging in mountain biking; no report of pain status At the end of treatment, chiseling pain had gone and had not come back at 3-month follow-up; pain in toes still there; coping better and ‘‘less jumpy’’

20 weekly hypnosis sessions: hypnotic suggestions of ‘‘sock and bandages’’ for left foot ‘‘acting as analgesia’’; ‘‘imagining being injected with pethidine’’; audiotape of hypnotic analgesic; self-hypnosis training after first 6 sessions; last 5 sessions focused on therapeutic effects, relapse prevention; psychoeducation and cognitive-behavioral therapy techniques for managing depression and anxiety and improving coping 64 hypnosis sessions over 3 years: 3 phases: a & b) suggested movements of ‘‘leg’’ and becoming normal size; c) suggested shrinking of phantom limb

Pain severity was reduced and daily onset of pain had been delayed to late at night; affective pain reduced significantly; increase in self-confidence, control over pain

Above-knee amputation of left leg (no pain before)— PLP for 25 years, pain felt as intermittent ‘‘burning’’ and

Outcome

At end of treatment, phantom limb changed to more relaxed shape and had disappeared for most of time, with intermittent

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Siegel (1979)

Problem

Yes—highly hypnotizable

Ersland et al. (1996)

No

Rosen et al. (2000)

Yes, Stanford Hypnotic Clinical Scale for adults (highly hypnotizable)

Rosen et al. (2000)

Yes, Stanford Hypnotic Clinical Scale for adults (moderately hypnotizable)

3 hypnosis sessions: relaxation, suggestion in hypnosis to relax and contract muscles in both legs; patient experienced free movement in ‘‘toes’’ and ‘‘leg’’; transfer of suggested numbness in hand to left ‘‘leg’’; last session of treatment audiotaped to listen to at home Hypnosis for part of rehabilitation program, number of sessions not specified: relaxation and hypnotic suggestions for finger movement and uncramping

At 2-week follow-up 50%-100% pain relief from self-suggestion, or by listening to hypnosis audiotape; considerable reduction in unpleasant sensations; less bothered by residual PLP and sleeps normally Reduction in PLP (not quantified); feeling of control made residual pain more tolerable

12 sessions over 6 months: cognitive/behavioral treatment with hypnosis; in hypnosis, imagined phantom in comfortable position or moving in a comfortable way; also imagined skiing, both arms moving in rhythm

Traumatic amputation of fingers on left hand (no pain before)— Severe pain in entire hand, ‘‘cutting’’ feeling in ‘‘phantom fingers’’ especially during uncomfortable movements

12 sessions over 6 months: cognitive/ behavioral treatment with hypnosis; in hypnosis, imagined phantom in comfortable position or moving in a comfortable way; also imagined pain area shrinking

Pain-free during first hypnosis session—lasted 1 day then pain returned intermittently; at the end of treatment, pain intensity down from 80 to 50; pain frequency reduced by 55%, shrinking of phantom limb reported At end of treatment, pain intensity was down from 40 to 20 and pain frequency reduced by 50%; these gains maintained at 2.5 years’ follow-up (Continued)

Phantom Limb Pain

LeBaron & Zeltzer (1996)

bursts of pain; overall pain had been reduced from 8 to 1 on a scale of 0-10

continuous dull pain (‘‘leg’’ and ‘‘foot’’ in uncomfortable posture and ‘‘leg’’ too short); Phantom ‘‘leg’’ shorter than the ‘‘healthy leg,’’ flexed at the knee, and big toe of the phantom limb extended Amputation of left leg (pain status before not reported)— PLP felt as:  ‘‘jerking’’ in ‘‘leg’’  ‘‘cracking’’ in ‘‘toes’’  ‘‘stabbing’’ pain in sole of phantom ‘‘foot’’ (‘‘like a thousand pins’’) Above-elbow amputation of right arm (pain status before not reported)— PLP for 18 months in ‘‘fingers’’ and ‘‘wrist’’ (absent ‘‘fingers’’ and ‘‘wrist’’ in uncomfortable posture) Traumatic amputation of right arm (no pain before)— PLP for 5 years: radiating heat pain in ‘‘arm’’ and ‘‘fingers’’; abnormal posture/contraction in ‘‘fingers’’ and ‘‘arm’’; felt fingers crumbling with cramping pain

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TABLE 1. Continued Study

Hypnotizability Assessed

Problem

Treatment

No

Avulsion of left brachial plexus (no pain before)— PLP for 5 years, pain felt as intermittent cramping in ‘‘denervated arm’’ and burning sensation; more frequent ‘‘shooting’’ (‘‘like small electric shocks’’) pains through arm; and ‘‘throbbing’’ in knuckles (‘‘hand’’ in uncomfortable ‘‘clenched’’ posture)

Previous experience of pain control and subjective movement of left hand in mirror apparatus; 1 session of hypnosis with suggestions of a return to the mirror experience and of age regression to time before the injury

Rickard (2005)

Yes, Stanford Hypnotic Clinical Scale for adults (moderate-high hypnotizability) No

20 patients with PLP: 19 lower-limb amputees and 1 upper-limb amputee

Participants were randomly assigned to the hypnosis treatment group (n ¼ 10) or control group (n ¼ 10): treatment group received individualized tailored hypnotic interventions; control group did not receive any treatment 6 weekly sessions of hypnosis treatment: sessions included visualization, guided imagery of patient’s choice (garden, beach, woods), and movement of phantom limb; relaxation and training self-hypnosis enabled them to continue it three times a day

Bamford (2006)

PLP in 25 amputees: 18 lower-limb amputees and 7 upper-limb amputees

During experience of moving left hand in hypnotic virtual ‘‘mirror’’ and during ageregression, PLP was lost; experience of left hand movement not as strong as in real mirror apparatus; pain was rated 4 before hypnosis, 0 during hypnotic mirror, and 2.5 after hypnosis on 10-point scale; long-term effect not reported At entry (time 1), both groups were found to be similar

Statistical analysis revealed significant change in pain scores at baseline, after treatment, and at follow-up for 22 cases (3 patients did not return questionnaires at 6-month follow-up)

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Oakley et al. (2002).

Outcome

TABLE 2. Outcome Studies Using the Cognitive-Behavioral and Relaxation Approach Study

n

Problem

Design

Treatment

Follow-Up

Single subject

Progressive muscle relaxation, EMG feedback, and reassurance

6 mo– 3 y

Dougherty (1980)

1

Above-knee amputation of left leg— continuous pain for 3 mo after surgery

Single-subject case report

2 sessions, each 45 min of EEG biofeedback training (discontinued after 2nd session by mutual agreement); EMG biofeedbackassisted relaxation training over 3 wk

5 wk

Belleggia & Birbaumer (2001)

1

Amputation of the right upper limb— Burning and shooting phantom pain

Single-subject case report

Training of EMG biofeedback followed by temperature training (6 sessions each); each session lasted for 30 min

At 3 mo and 12 mo

Intractable phantom limb pain

Randomized

5 patients received 10 daily 90-min sessions (over a duration of 2 wk) of feedback-guided sensory training, in which they had to

3 mo

Flor et al. (2001)

10

At follow-up, 8 of the 14 chronic patients had substantial pain decreases; 2 recent amputees showed complete relief from pain. Patients’ anxiety levels significantly reduced from a posttreatment average of 72  15 to 24  23 (t ¼ 6.93; p > .001), and the average pain levels (pain ratings 0 [no pain] to 100 [unbearable pain]) changed from 76  13 to 13  19 (t ¼ 11.17; p > .001). Although EEG biofeedback was ineffective, EMG biofeedback-assisted relaxation allowed the patient to greatly reduce the frequency and intensity of pain after 4 h of practice. However, at follow-up, pain returned. Baseline pain intensity (10-cm VAS) ¼ 3. At 3-mo follow-up, pain intensity decreased (VAS ¼ 0), and at 12 mo, no pain. Phantom limb pain (as measured with West Haven–Yale Multidimensional Pain Inventory) decreased significantly from (Continued )

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14 lower-limb amputees (above-knee ¼ 11; below-knee ¼ 3), 2 upper-limb amputees

Phantom Limb Pain

16

Sherman et al. (1979)

Results

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TABLE 2. Continued Study

n

Problem

Design

Treatment

Follow-Up

discriminate the frequency and location of high-intensity nonpainful electric stimuli through 8 electrodes attached to the stump; control group of 5 patients received standard medical treatment (analgesic medicine, transcutaneous nerve stimulation, physical therapy)

9

5 patients had lower-limb amputation and 4 had upper-limb amputations

Single subject

7 sessions of thermal/ autogenic biofeedback over 4-6 weeks; psychometrics: Beck Depression Inventory, State Trait Anxiety Inventory

6 wk

pretraining to posttraining (t[4] ¼ 3.27; p ¼ .008) as well as from pretraining to follow-up (t[4] ¼ 4.85; p ¼ .002) in experimental group; no significant changes in control. Cortical reorganization (measured as the distance between the lip locations on the right and left hemispheres) was reduced in the training (t[4] ¼ 1.75; p ¼ .07 posttraining; t[4] ¼ 2.00; p ¼ .05 at follow-up) but not in the control group. Slight changes in BDI and STAI scores but not statistically significant. Pain intensity measured by VAS at baseline was (38.2  21.4). A 20% pain reduction seen in 5 of the 9 patients in weeks after session 4, and $30% pain reduction (range 25%66%) seen in 6 out of 7 patients in the weeks following session 6. VAS for week 7: 16.8  6.3.

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Harden et al. (2005)

Results

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guidelines (Hawkins, 2001). Additionally, a review of randomized controlled studies suggested that hypnosis has a reliable and significant impact on acute procedural pain and chronic pain (Patterson & Jensen, 2003). Moreover, evidence from recent neuroimaging studies have shown that changes in the subjective experience of pain produced by suggestions given in hypnosis are reflected in alterations in the activation of brain areas known to be involved in normal pain perception (Ersland, Rosen, Lundervold, Smievoll, Tillung, Sunderberg, & Hugdahl, 1996; Rosen, Willoch, Bartenstein, Berner, & Rosjo, 2000). Despite the large number of studies on pain and hypnosis, only a few focus on PLP. Hypnosis was one of the treatment strategies identified by Sherman et al. in a survey of treatments used to manage PLP in the USA (Sherman, Sherman, & Gall, 1980). Unfortunately, fewer than 1% of phantom limb patients who had sought treatment were treated by hypnosis and there was no information reported regarding the duration or type of hypnotic interventions. The literature search carried out during the present review revealed a dearth of systematic studies conducted to evaluate the effectiveness of hypnosis as a treatment for PLP. However, there are single case studies where hypnosis has been used to treat PLP, which provide insight into possible treatment strategies. These studies are summarized in Table 1. The results of the reviewed studies demonstrate the consistent effect of hypnosis on the reduction of PLP. PLP as significantly reduced in a majority of the amputees described in these studies. Suggestions used for shrinkage of phantom limb during hypnotic intervention in two cases (case 3 and 7) resulted in successful shrinkage of phantom limb; shrinkage occurred spontaneously in one case (case 10). It has been suggested that the phantom limb should be treated as ‘‘real’’ and the same pain management strategies applied as with a physically present limb (Oakley, Gracey-Whitman, & Halligan, 2002). Many patients with phantom limb sensations and PLP specifically say that they expect psychologic treatment to be directed at their phantom limb and not to the amputated stump (Oakley, Gracey-Whitman, & Halligan, 2002). For example, in one study, hypnotic glove anesthesia applied to the stump produced only temporary alleviation of the ‘‘burning’’ PLP; a later suggestion to visualize ‘‘a stream of cooling anesthetic agent’’ coursing through the amputated leg produced long-term pain reduction (Siegel, 1979). There are several important methodologic issues that make firm conclusions regarding the efficacy of hypnosis, specifically in the treatment of PLP, difficult. A majority of the studies in this review are single case studies. The case studies might provide insight into

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a detailed examination of the application of hypnosis in individual cases, but there are no published randomized controlled trials to examine the efficacy of hypnotic interventions in treating PLP. There is just one study (Bamford, 2006) that had a large number of patients, but there was no control population. A study conducted by Rickard (2005) found the hypnotic interventions to be effective in the management of stump pain and PLP (Rickard, 2005). The random assignment and comparable control group add to the reliability of the result of that study. However, it is difficult to make generalizations from these studies. The hypnotic interventions also vary widely from study to study. The hypnotic interventions included hypnotic imagery, hypnotic suggestions of shrinkage or movement of phantom limb, visualization and guided imagery. Self-hypnosis training was also provided to the patients in some of the studies. Only a few studies clearly described the hypnotic interventions and the status of the pain before the intervention. There is clearly a need for more randomized controlled trials that include larger samples and well defined standardized hypnotic interventions. A second issue is the association between hypnotic pain reduction and hypnotic suggestibility as measured by hypnotizability scales. Montgomery, DuHamel, and Redd (2000) found suggestibility to be an important variable in their meta-analysis. They reported that patients scoring moderate to high on hypnotizability scales are more likely to benefit from the hypnotic suggestions. Hypnotizability has been measured in only three of the cases reviewed here, and it is significant that in two of these three reports hypnotizability was found to be high. It has also been reported that people can increase their suggestibility with training and practice (Patterson & Jensen, 2003). It would be useful to measure suggestibility, as this might be an important issue in selecting patients for hypnosis-based PLP treatment who can respond readily and can also indicate those that might need additional training or support. Cognitive and Behavioral Approaches The cognitive approach sees pain as an experience mediated by cognitions. From the cognitive-behavioral perspective, people with pain have negative expectations about their own ability to control certain motor skills. Such negative and maladaptive appraisals about the situation and self-efficacy may lead to demoralization, inactivity, and overreaction to nociceptive stimulation (Turk & Gatchel, 2002). Regardless of the origin of the pain, a cognitive-behavioral approach to treatment of chronic pain assumes that modifying cognitions might reduce pain levels and provide the

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TABLE 3. Studies Using Mirror/Visual Feedback (MVF) Study

n

Problem

McLachlan et al. (2004)

1

Lower limb amputation

Single case study

Brodie et al. (2007)

80

Upper-limb amputees

Randomized controlled study

Single-blind randomized

Treatment

Follow-Up

Not Exercise of intact limb in specified front of full-length mirror (positioned so as to provide patient the full reflection of his intact leg) twice a day for 2 weeks. In third week patient performed exercises without mirror. All aspects of treatment did not vary during MVF treatment. Two conditions. In both conditions, subjects attempted 10 movements, each repeated 10 times, with both phantom and intact limb. Mirror therapy (n ¼ 41): subjects placed their intact limb into the mirror box and align their phantom with the image of intact limb. Control group (n ¼ 39): subjects aligned their intact leg and phantom leg to either side of the mirror while it was obscured. 6 month Patients randomized to graded motor imagery program (experimental group; n ¼ 25) and standard medical and physiotherapy care (control group; n ¼ 26). Treatment over 2 weeks

Results Phantom pain intensity: (pre/post: 5-9 out of 10/0 out of 10). Stump Pain: (pre/post: 0-2/1). Control over phantom leg: pre/post 0%/25%-30% (none at all ¼ 0% to complete ¼ 100%).

Both conditions resulted in significant attenuation of phantom sensations and phantom limb pain, but the mirror condition did not provide additional benefits over the control condition (motor imagery without mirror).

Significant decrease in pain and function for the motor imagery group compared with the control group. Mean (95% CI) decrease in pain between pre- and posttreatment (100-mm VAS scale): 23.4 mm (16.2-30.4 mm) for motor imagery group; 10.5 mm

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Clinical assessment of Moseley (2006) 51 (complex phantom limb patients regional pain found hyperalgesia and syndrome ¼ 37; allodynia of the stump, brachial plexus swelling, and avulsion temperature changes injury ¼ 5; amputees ¼ 9)

Design

Chan et al. (2007)

22

Phantom Limb Pain

Sumitani et al. (2008)

22 patients

(1.9-19.2 mm) for control group. Not Baseline median VAS M group performed Phantom limb pain after Randomized specified scores were similar in all movements with foot or leg amputation sham-controlled. groups: M, 3.1 cm (range amputated limb in front 3 treatment groups: 1.5-9.3); CM, 3.8 cm of a mirror. mirror (M; n ¼ 6); (1.8-7.0); MV, 2.7 cm CM group performed covered mirror (2.2-6.3); p ¼ .62. movements with both (CM; n ¼ 6); mental Mirror therapy was found to intact and amputated visualization (MV; be effective compared limbs when an opaque n ¼ 6) with covered mirror or sheet covered the mirror. mental visualization MV group closed eyes and therapies in reducing performed movements phantom limb pain. After with amputated limb. 1 month of therapy, Each group performed their M group differed assigned therapy under significantly from CM direct observation for 15 group (p ¼ .04) and MV minutes daily for 1 group month. (p ¼ .002). Pain intensity across all MVF: exercise of intact Pilot study. 11 single-limb amputees patients was significantly limb facing the mirror (trauma: n ¼ 5; malignant No control over reduced (numeric rating and imagining medicine intake tumor: n ¼ 6); brachial scale [NRS]: pre 6.6  movements of affected/ and other plexus avulsion injury: n 1.7; post 4.2  2.8; p < amputated limb)—10 conventional ¼ 7; traumatic peripheral .002). minutes once a day for treatment. nerve lesions: n ¼ 2; Patients with willed a period of 20.4  23.8 spinal cord injury: n ¼ 2 visuomotor imagery weeks reported significantly lower pain intensity (NRS: pre 6.3  1.5; post 3.2  2.4; p < 0.001), whereas patients without visuomotor imagery did not (NRS: pre 7.3  2.0; post 6.4  2.4; p ¼ .50).

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patient with a better method of managing pain. A systematic review conducted by Morley, Eccleston, and Williams (1999) concluded that psychologic treatments of chronic pain based on the principles of cognitive and behavioral therapy are effective. There is also evidence for efficacy of cognitive and behavioral therapy for treatment of neuropathic pain (Wetering, Lemmens, Nieboer, & Huijsman, 2010). Biofeedback and Other Behavioral Methods. Whenever the body is injured, the tendency is to tense the muscles in that body aream thereby immobilizing the site to further trauma. In the case of acute pain this response has obvious value, but when the muscles are chronically tensed this tension itself produces more pain, which in turn causes more tensing. A pain-tension cycle begins, which is compounded by other factors, e.g., lack of sleep, depression, and lowered activity (Linton, 1982). Therefore, behavioral methods such as biofeedback, relaxation, and imagery could help to relieve the muscle tension. Peripheral feedback, such as electromyographic (EMG) biofeedback, is well established as a treatment of chronic back pain and chronic headaches. The use of biofeedback to treat PLP is not novel, and usefulness of some of these behavioral therapies in managing PLP has been assessed recently in a few studies. Various studies using EMG biofeedback, sensory discrimination training, and other relaxation techniques for the management of PLP are summarized in Table 2. The studies using EMG biofeedback or relaxation technique reported decreases in PLP. Taken as a group, the data suggest that patients with PLP may benefit from relaxation and biofeedback treatment. A lack of data from well controlled studies makes this conclusion somewhat tentative. Most of the studies in this group used a rather weak one-group pre-/posttest design. There are several problems (e.g., controlling for history, reactivity to measurement) with this design which makes causal inference difficult. Also, extraneous factors such as placebo, attention, and demand characteristics can not be accounted for in a one-group design. Even if statistically significant differences between pre- and posttest are found, it is difficult to attribute the change to specific treatment unless there is a convincing control condition. Only one study had a control condition of any type (Flor, Denke, Schaefer, & Grusser, 2001). Another limitation of these studies has been the lack of a broad range of objective outcome measures. The typical investigation had EMG and pain ratings, but few reported on other pain-related behaviors, such as reduced activity and sleep difficulties. Studies used different measurement tools to assess pain, and the small sample sizes further limit generalization.

One advantage of relaxation and other cognitivebehavioral therapies is that they are relatively inexpensive and easy to administer. Behavioral techniques such as EMG biofeedback, skin temperature feedback, and muscle relaxation procedures allow monitoring of bodily responses and thereby indirectly modify sympathetic activity (Freedman, 1991). Distraction may also be involved during the process of behavioral interventions. Morgenstern (1964) found that when patients with PLP concentrated on performing several sensory-motor tasks in the presence of sensory distractions in daily sessions for a period of 3 months, PLP was substantially reduced. Biofeedback facilitates cognitive changes in patients by demonstrating that physiologic processes are under their control and that they are in fact able to influence these physiologic processes (i.e., enhancing selfefficacy) (Turk & Gatchel, 2002). Thus, biofeedback may simply be a vehicle to shift the patient’s attention from external to internal methods of pain coping, making perceived pain more tolerable (Linton, 1982). It would be helpful if future studies using such interventions assess changes in perceived control and selfefficacy in the treatment of PLP. Visual Imagery and Mirror Therapy. Recently, studies have reported the potential of visual feedback in the treatment of a variety of pathologic pain conditions (McCabe, Haigh, Ring, Halligan, Wall, & Blake, 2003; Moseley, 2006). Ramachandran, Brang, and McGeogh (2009) have used the mirror box to provide visual feedback to upper limb amputees. They found that when the reflection of a moving normal limb in a mirror is superimposed on the felt position of the phantom limb, it can modify the experience of phantom limb, and the associated pain was relieved when the patients subsequently exercised the normal limb. They suggest that mirror therapy helps PLP by resolving visual-proprioceptive dissociation in the brain. Studies using visual feedback and mirror therapy in the management of PLP are summarized in Table 3. A randomized controlled trial in 18 patients showed significant benefit of mirror therapy in PLP patients when compared with two control groups (Chan et al., 2007). That study report lacked a description of patient characteristics, and follow-up data were not provided. Moseley (2006) reported an average 23% decrease in pain on a 100-mm visual analog scale (VAS) after 6 weeks of graded motor imagery and a 32% decrease in pain at 6 month’s follow-up. In that study, participant results were pooled regardless of the cause of pain (complex regional pain syndrome 1, brachial avulsion, or amputation), thereby making it difficult to evaluate the efficacy of mirror therapy in PLP

Average 2.8 y (range 26-40 mo) Case series

5

10

Schneider et al. (2008)

de Roos et al. (2010)

One-side leg amputation ¼ 9; both-leg amputation ¼ 1

3-15 sessions of EMDR

Case series

12-24 mo

4 of the 5 clients completed the prescribed treatment and reported that pain was completely eliminated or reduced to a negligible level (1 out of 10). Complete elimination of PLP in 2 cases and decrease of pain in 3 cases (of 4 patients, numeric rating scale pre/post: 9.5/2) and remained at the same level at follow-up. Substantial reduction in chronic PLP in 8 participants. 4 were completely pain free at 3 mo follow-up; 3 of 6 available participants were completely pain free. Follow-up at 1 y (only 1 case) and at 3 y (only 1 case) Case series 3-9 sessions 5 Wilensky (2006)

Foot amputation ¼ 2; leg amputation ¼ 1; finger amputation ¼ 1; arm amputation ¼ 1 4 lower-limb amputees; 1 upper-limb amputee

Follow-Up Design Treatment Problem n Study

TABLE 4. Summary of Studies Using Eye Movement Desensitization and Reprocessing (EMDR)

Results

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patients. However, another controlled trial found mirror therapy to be no better in the reduction of PLP compared with motor imagery without a mirror (Brodie, Whyte, & Niven, 2007). That study showed that patients in both the groups experienced significant attenuation of phantom sensations and pain. However, patients in mirror therapy group could experience the willed visuomotor imagery of their phantom limb more frequently than those viewing the intact limb movements alone (Brodie, Whyte, & Niven, 2007). A recent systematic review of three randomized controlled studies found limited evidence of mirror therapy in reducing PLP and has suggested the need for more rigorous controlled studies to investigate the analgesic efficacy of this therapy (Seidel, Kasprian, Sycha, & Auff, 2009). Eye Movement Desensitization and Reprocessing. Eye movement desensitization and reprocessing (EMDR) is an evidence-based psychotherapeutic approach that has proved to be effective in the treatment of psychologic trauma. The theoretical framework, adaptive information processing (AIP), on which this psychotherapy is based illustrates the broad application of the treatment. According to this model, a high level of disturbance at the time of a traumatic event causes the information-processing system to fail, making it difficult to properly assimilate the experience into the normal comprehensive memory networks. As a result, these unprocessed memories are stored in isolation and contain the affect, thoughts, sensations, and behavioral responses that were encoded at the time of the event (Schneider, Hofmann, Rost, & Shapiro, 2007; Shaprio, 2001). The AIP model suggests that many forms of chronic pain, including PLP, are a result of inappropriately stored somatic memories related to the traumatic event. These memories are thought to be a major factor in the maintenance of PLP. The reprocessing of these physiologic encoded memories may help to reduce PLP. The literature search yielded two single case studies (Russell, 2008; Schneider, Hoffmann, Rost, & Shaprio, 2007) and three case series (de Roos, Veenstra, de Jongh, den Hollander–Gijsman, van der Wee, Zitman, . van Rood, 2010; Schneider, Hoffmann, Rost, & Shapiro, 2008; Wilensky, 2006). These studies evaluating the role of EMDR in the management of PLP are summarized in Table 4. There were no randomized controlled studies in the literature evaluating the effectiveness of EMDR in PLP. The results of the reviewed case reports and case series suggest that EMDR may be useful in the treatment of PLP. One of the larger series evaluating the effectiveness of EMDR (de Roos et al., 2010) showed a significant attenuation of PLP in six out of ten patients, which was

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maintained at long-term follow-up (26-40 months). This report suggested that EMDR is effective in not only alleviating PLP but also reducing stress and depression levels. Further rigorous studies with larger sample sizes and randomized designs are warranted to confirm these findings.

CONCLUSION Phantom limb pain is a complex entity, which has historically proved to be difficult to treat. Medical management of PLP has had very limited success. This has spawned interest in nonpharmacologic interventions for the treatment of PLP. The aim of the present review

was to examine the role of psychologic factors in the causation of PLP and to evaluate the effectiveness of the various psychologic therapies in the management of this condition. The review of pathophysiologic factors indicates that the psychologic factors do not seem to play a role in the origin of PLP but may exacerbate and affect the course and severity of the phenomena. Although the studies on the treatment of PLP reviewed here have reported various levels of success in the management of PLP, it is difficult to make firm conclusions regarding the efficacy of these interventions, because there is a dearth of controlled studies of psychologic interventions specifically in the treatment of PLP.

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Phantom limb pain and its psychologic management: a critical review.

Phantom limb pain is a puzzling phenomenon, from the viewpoints of both the patient experiencing it and the clinician trying to treat it. This review ...
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