Etiopathogenetic Explanations and Treatment Options in Phantom Limb Syndrome. A Literature Review

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ScienceDirect Procedia - Social and Behavioral Sciences 187 (2015) 307 – 311

PSIWORLD 2014

Etiopathogenetic explanations and treatment options in phantom limb syndrome. A literature review Andreea Rodica Ardelean*, Ion Dafinoiu Alexandu Ioan Cuza University of Iasi, nr.3, Toma Cozma Street, 700554, Iasi, Romania

Abstract Phantom limb syndrome is a controversial subject when talking about its etiopathogenetic mechanisms and efficient treatment modalities. The article reviews different etiology mechanisms, both neurological and psychological, as there is no consensus among specialists in these fields, as well as different ways of dealing with reducing pain in people that suffer from phantom limb syndrome. It also highlights the gasps on the subject and the need for future research on certain aspects of the phenomenon. © 2015The TheAuthors. Authors.Published Published Elsevier © 2015 byby Elsevier Ltd.Ltd. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). Peer-review under responsibility of the Scientific Committee of PSIWORLD 2014. Peer-review under responsibility of the Scientific Committee of PSIWORLD 2014. Keywords: phantom limb; etiology explanations; neuromatrix theory; treatment; mirror box; hypnosis

1. Introduction Phantom limb syndrome is defined as the persistence of the perception of a limb that has been amputated and has been mentioned starting with Antiquity. The one who firstly mentioned it in medical literature and offered it a scientific character was the neurologist Silas Weir Mitchell in 1871. Since then, scientists in neurology and psychology have been stating different etiologies and treatment modalities, none of them being agreed upon as most efficient. The present study reviews theoretical articles and studies on etiology and treatment of this syndrome.

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Corresponding author. E-mail address: [email protected]

1877-0428 © 2015 The Authors. Published by Elsevier Ltd. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). Peer-review under responsibility of the Scientific Committee of PSIWORLD 2014. doi:10.1016/j.sbspro.2015.03.057

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2. Neurological theories regarding phantom limb syndrome etiology The first explanatory theory is the peripheral theory, which underlines the importance of neuromas. According to this theory they send impulses through the spinal cord to the brain (cortex), impulses that may be felt as painful. Thus the neurons that were sectioned and that are located in the stump create, by unusual firing, sensitive messages in the somatosensory cortex. Even though they are not well organized and clearly defined, they entertain and maintain the perception of the missing part of the body, as well as the pain located in that corporal segment (Sherman & Sherman, 1989; Devor & Faulkner, 1999). This first theory of etiopathogenesis stands for some therapies based on the administration of analgesics, as well as for the use of massage and the electrical stimulation of the stump (Sherman & Sherman, 1989). Because “the phantom” did not disappear specialists have also considered surgical interventions, but they did not provide a significant amelioration.The peripheral theory has been contested frequently, for some arguments like the fact that the phantom limb syndrome appears in many cases immediately after amputation, while the neuroma formation is a process that requires some time post amputation, it is not an immediate process (Wall & Fitzgerald, 1981). Another argument against the peripheral theory would be the fact that the PLP phenomenon is also present in the case of spinal traumatisms, and in this case the peripheral nervous system is not at all affected (Hotz-Boendermaker et al, 2008; Curt, Yengue, Hilti, Brugger, 2010). In order to demonstrate the peripheral origin of phantom limb syndrome, it has been stated that it does not appear in the case of children who are congenitally missing a limb. But this hypothesis has been proved to be false, because some patients with congenital absence of a limb reported to have lived the sensation of the presence of the missing limb (Melzack, 1996). A study conducted by Wilkins, McGrath, Finley and Katz (1998) has reported the presence of this sensation in 42% of those who presented a congenital absence of a limb, 29% of them suffering also from phantom limb pain. In contrast to peripheral theories, central theories claim that phantom limb syndrome is the result of the firing of central nervous system neurons. The loss of afferent input determines the firing of neurons situated in the central nervous system, and it provokes permanent changes in the synaptic structure. This results in an increase of nerves irritability and a reduction of the inhibitory process, this phenomenon being difficult to interrupt. Cortical reorganization is one of the most cited explanations in the phantom limb syndrome etiology. Literature suggests that the cortical area corresponding to the amputated part of the body is “taken in responsibility” by the representative zones next to that area, from the somatosensory cortex, but also from the motor cortex (Melzack, 1992; Melzack, 2005; Moseley, 2006). Ronald Melzack (1992) introduced the term “the neuromatrix theory” to explain the sensations felt in the amputated limb, as phantom limb sensation and phantom limb pain. The neuromatrix theory follows naturally after peripheral and central theories, integrating elements from both of them. This theory sustains the idea of a neuronal matrix that integrates different types of inputs generated from different body parts, including somatosensory, limbic, visual, thalami-cortical elements. The neuromatrix would contain three important pain dimensions: sensorial, cognitive and affective. The internal body conscience is created at the cerebral level, being activated by perceptual impulses. The term “neurosignature” was proposed by Melzack to refer to the activity pattern generated by the brain, which is actualized by the consciousness and by the body and self -perception. Melzack states that phantom pain is generated by deprivation of many inputs from limbs to the neuromatrix, causing the formation of abnormal signature. The central theory of phantom limb syndrome origin is sustained by an experiment made by Hinedenmayer. He realized a study where participants that suffered from phantom limb sensation had to perform the same tasks with the intact limb, as well as with the “missing” limb. He examined EEG results and they showed “the same modifications for both body parts” (Hindenmayer, 1962).

3. Psychological theories regarding phantom limb syndrome origin Paralel to neurological theories, phantom limb syndrome has been considered to be a phenomenon of psychological origin. Difficult psychological adaptation to a new medical situation and body change, the change in the quality of live, has been considered to stand for the origin of phantom sensations and their maintainence in time (Weil, 1991). This theory is based upon the presumption that the phantom limb constitutes the object of narcissistic

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desire to maintain body integrity in front of the violent and imminent loss of a part of the self (Dejours, 2001). The ideea that the phantom limb constitutes a denial of the limb loss has persisted in scientific writings (Kolb, 1954, apud Raffin Etelle, 2011). On the other hand, there are many studies that state the absence of significant correlations between phantom limb sensations and coping modalities or scores obtained by subjects to different emotional disturbances (as depression or anxiety) (Fisher & Hanspal, 1998). Even if psychological factors didn’t have an essential contribution in phantom limb genesis, they certainly have an influence on the way people who suffered an amputation adapt to postamputation life and on the manifestations specific to the phenomenon (the level of perceived pain, for example), according to the biopsychosocial model of pain. Kolb (1954) has conducted a study on the influence of psychological factors on the phantom limb syndrome. The results showed that there is no specific personality structure that correlates with phantom limb presence, but there are some dominant personality traits resembling those proposed by Engel in 1959, and that were called „the patient susceptible to pain”, patient that presents some traits like hostility and autopuntition, that play an important role in maintaining chronic pain. As mentioned above, there are some studies that underlined the importance of psychological factors in the PLS genesis and evolution. Parkes (1973), in a study realized on 46 patients, has found a positive correlation between the persistence of postamputation pain and two personality traits: rigidity (resistance to changes) and compulsive self-trust, which consists in wanting to assume all responsibility, even when others show disponibility to help you. The presence of these traits can be a predictor for phantom limb pain. Social support is frequently studied when investigating chronic pain, so another important factor that seems to influence the adaptation to phantom limb pain is the control over this pain. Persons who have an internal locus of control seem to have a lower intensity of pain, acording to some studies (Crisson & Keefe, 1988 apud Curelli, 2004), and they are less depressive (Skevington, 1993; Keefe & Williams, 1990 apud Curelli, 2004). A study conducted by Tippens in 1992 (apud Curelli, 2004), presents some factors that intensify and have the tendency to chronicize the pain in the missing limb: anxiety because of physical and psychological stress, endogene or reactional depression, hysterical reaction to limb loss, the need for pain, higher degree of hostilty. The subject about emotional disturbances and phantom limb pain is controversial, as other authors (Katz, 1992 Melzack, 1992), studying a group of amputated people for a period of more than 2 years, argue that there is no correlation between PLP and despressive or anxious persons. Sherman (1989), in a study on amputated people, shows that 37% of them present an intensification of phantom limb pain after a stressful event, but also a significant growth of stress level because of the existence and persistence of phantom limb pain (in 44% of them). . Pucher, Kickinger &Frischenschlager (1999) have found a significant correlation between some coping strategies and the pain felt in the phatom limb. Hill, Niven & Knussen (1993) have found a significant correlation between the coping strategy called „dramatizing” and PLP, as well as with psychological distress. The same authors have found in a consecutive study to the one mentiones above, that „dramatizing” as a copying strategy is a predictor not only for the severity of phatom pain, but for perceived physical incapacity and psychological distress (26%, 11% and 22% of variance). As we have seen, previous studies presented some psychological factors that are in relation with PLP (such as stress, anxiety, depression, cognitive factors as coping strategies or locus of control), but it remains the open question: are they in direct correlation or mediated by othe variables? 4. Treatment options for phantom limb pain Treatment for phantom limb pain is a controversial subject, on one part because studies on this theme are limited, on the other part because there is no general consensus over optimal intervention in phantom limb syndrome. In the case of pharmacological treatment, studies have shown that even though doctors consider that medication (analgesics, antiinflammatory medication) is efficient in reducing pain, but only 10% of the patients suffering from phantom limb pain admit its efficiency. The opioids are considered to be the most efficient in completing the effects of analgesics and local anesthesia. Generally, analgesics that are administered pre and post-operatory have a benefic effect on the phantom limb pain, moderating even its appearance (according to the theory of „pain memory”, it might be a causal factor for phantom limb pain (Chapman, 2010, Middleton 2003, Flor, Nikolajsen & Jensen, 2006). Some studies, on one part, sustain the efficacy of pharmacological interventions (Bach, Noreng & Tjéllden, 1988;

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Schug, Burrell , Payne & Tester, 1995) while on the other part, there are studies that deny the existence of any amelioration of pain sensation in the phantom limb (Pinzur, Garla, Pluth & Vrbos (1996); Nikolajsen, Ilkjaer, Christensen, Krøner & Jensen, 1997). Some authors sustain also the efficacy of antidepressants (amytriptiline especially) or anticonvulsants (Chapman, 2010; Bone, Critchley & Buggy, 2002; Traub, 2012). Acupuncture has proved to be an efficient way of treatment, especially in regions where it is traditionally used as alternative medicine (Tseng, Chen, Lee, 2014; Bradbrook, 2004) but most of the studies are case studies. Among most utilized treatment methods we can cite mirror therapy and virtual therapy. Of all the psychological therapies, cognitive-behavioural therapy and hipnotic interventions had a certain impact in reducing pain in phantom limb. The temporary reduction of pain in the phantom limb has been also obtained at the cerebral level, by stimulating ventral zones of the brain and also the caudate nucleus. As we have stated before, one of the most utilized treatment modalitiy is the mirror box therapy, mentioned firstly by Ramachandran and Rogers-Ramachandran (1996). The patient can see the intact limb’s reflection with the help of a mirror parasagitally placed in a box, between feet or arms, and he can „move” like this, the missing limb. The virtually limb in the mirror coincides with the phantom limb. After weeks of treatment, patients felt an amelioration of cramps and the sensation of „ freezed limb” has disappeared. The efficacy of this type of intervention seems to be quite high, about 60% (9/15 upper limb amputees) (Ramachandran & Rogers-Ramachandran, 1996). Even if it solves “the cause” that Ramachandran supposed to be at the basis of the syndrome - the inadequacy of motor and visual signals - the efficacy of this treatment is not fully explained. Scientists ascertain that if this type of treatment is used along with imagary exercises, its efficiency is higher (Chan, Witt & Charrow, 2007). Relaxation techniques and hypnosis are frequently mentioned in literature (Beaumont, Mercier, Michon, Malouin & Jackson, 2011; Harden, 2010). Relaxation techniques followed or not by biofeedback or hypnotic suggestion, have proved to be efficient in reducing phantom limb pain. The main reason is that they interrupt the circle painanxiety-tension (12 out of 14 patients have stated the significant reduction of phantom limb pain, according to Manchikanti & Singh (2004). There are not so many quantitative studies to sustain the superiority of psychological treatments over other types of treatment, but many case studies have described a significant reduction of phantom limb pain with the help of neurofeedback, relaxation techniques and hypnosis. A case study utilizing guided imagery and neurofeedback demonstrates the efficacy of these interventions in reducing pain, even after 12 months of follow-up. Another study, realized by Belleggia & Birbaumer in 2001, asserts that 45% of the participants that used hypnosis have noticed a significant reduction of PLP, a successful technique being “hand analgesia”. Nevertheless, 35% of the patients have reported a relapse of pain at the interruption of the hypnotic treatment. 5. Conclusions. To conclude, the multidisciplinary perspective of this phenomenon, even though it gets wider in the last two decades, is still in deficit of consensus concerning aetiology and treatment, and it needs randomized clinical studies, as literature presents mainly case studies especially when investigating treatments. Future research using control groups and placebo controlled trials is needed for a better understanding of the most efficient treatment option and a completion of scientific information that we have at present.

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