2015; *: 16:**–** 761–764 Pain Medicine 2014; Wiley Periodicals, Inc.

Case ReportPeripheral Neuromodulation for Successful Successful Peripheral Phantom Limb Pain Neuromodulation for Phantom Limb Pain

Philip Cornish, BHB, MBChB, FANZCA, FFPMANZCA and Cindy Wall, BPsych(Hons), MPsych(Clin), PhD, FAPS Specialised Pain Medicine Pty Ltd, Adelaide, South Australia, Australia Reprint requests to: Philip Cornish, BHB, MBChB, FANZCA, FFPMANZCA, Specialised Pain Medicine Pty Ltd, 2 Mary Street, Unley, Adelaide, SA 5061, Australia. Tel: + 61870706750; Fax: + 6181256551; E-mail: [email protected]. Disclosure: The authors do not have any conflicts of interest.

Abstract Setting. For decades, the heterogeneity of the amputee population and the complex interaction of biopsychosocial factors have confounded researchers’ attempts to develop an effective treatment for phantom limb pain. Therefore, it remains difficult to treat, and affected patients often experience decreased quality of life, increased psychological distress, and poorer health outcomes. Patient. In the case study, we report a novel strategy for the peripheral placement of neuromodulation leads for the treatment of phantom limb pain in a patient who subsequently described complete and consistent pain relief independent of significant variations in psychosocial stress. Key Words. Phantom Limb Pain; Neuromodulation; Peripheral Field Stimulation

Effective pain relief for amputees with phantom limb pain (PLP) represents a unique challenge in pain medicine. It is estimated that as many as 80% of amputees experience neurological sequelae in the form of phantom sensation, residual limb pain, or PLP [1–4]. Postamputation pain results in significant disability, psychological distress,

impaired work capacity, decreased quality of life, and additional related morbidity [5–9]. Despite the variety of therapeutic modalities employed to alleviate symptoms, as few as 10% of patients report long-term relief [10–13]. Amputees experiencing moderate to severe PLP frequently report that postamputation pain has a greater impact on their lives than the amputation of the limb itself [7–9]. Central maintenance of PLP has been a widely accepted theorem for some time [14–16]. Approaches to pain management of PLP have thus tended to reflect this theorem, targeting structures at varying levels of the central sensory pathways (e.g., mirror box therapy [17,18] and psychological treatment [19] at supratentorial level, dorsal column stimulation at spinal cord level [20–23]). Results from these therapeutic approaches have however been variable and indeed were comparable to contemporaneous investigations into peripheral nerve stimulation [24–26] and more recent studies using continuous peripheral nerve blocks [27–29], peripheral nerve stimulation [30–33], and pulsed radiofrequency of the peripheral nerves [34]. In this case study, we report an innovative approach to the placement of neuromodulation leads for management of PLP following amputation. We describe the successful implantation of neuromodulation leads into the stump of a below knee amputation in a middle-aged woman with severe PLP and discuss the treatment and theoretical implications of this novel procedure. History and Presentation The patient, a 40-year-old elite sportswoman had undergone right below knee amputation 2 years earlier on a background of chronic regional pain syndrome of the right ankle following recurrent fractures of the ankle, persistent pain, and a subsequent unsuccessful fusion of the joint. She had requested amputation as she felt her greatest chance of walking again was via a fitted prosthesis. Consultations with pain specialists and psychologists concluded that there were no other more conservative options available and that her psychological profile supported her ability to make a rational decision and to recover from the surgery. The below knee amputation was accompanied by severe deafferentation pain, including PLP in the ‘ankle’ (crushing sensation), electrical jolt-like spasms in the right leg (up to 50/day), and allodynia in the skin covering the stump

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Cornish and Wall (unable to bear the stump to be touched). She proved intolerant to tricyclic antidepressants, gabapentinoids, and opioids. Despite her distress, she remained determined to walk with the aid of a prosthesis which led to her requiring revision of the soft tissues around the stump to aid a better fit. This procedure resulted in amplification of her pain experience, and continuous peripheral nerve catheters (sciatic and femoral) were placed to gain temporary control of her pain. Intravenous calcitonin was then administered as a daily infusion of 100 IU on three successive days, but with cessation of the peripheral nerve catheters, there was no alteration in her pain. A trial of dorsal column stimulation was then organized. Two leads were placed in the epidural space and an extensive on-table trial conducted at varying levels between T8 and T10, with stimulation being achieved in the left leg and back but not in the affected right leg. After further discussion postprocedure, it was decided to revisit the option of neuromodulation but to place the leads in the subcutaneous tissues on either side of the stump, targeting the allodynic area which extended right across the stump. There was to be no attempt to target specific nerves. Lead placement was subsequently achieved under regional anesthesia (sciatic −15 mL 1% Ropivacaine; femoral −10 mL 1% Ropivacaine; nerve blocks using nerve stimulator guidance, 0.5 mA, 2 Hz) with the leads (‘linear 3–6 8 contact’ leads, Boston Scientific, Marlborough, MA, USA) placed in the medial and lateral aspect of the stump under ultrasound guidance to ensure that they lay within the adipose tissues (Figure 1). Once the regional blocks had regressed, stimulation was instituted, and there was rapid diminution of all three components of the patient’s pain to the point that they were no longer present. This effect lasted the entire duration of the 10-day trial period. As an incidental finding, the patient noted that on mobilizing, she had difficulties with balancing on her left leg, and it took a couple of days to readjust to standing upright. She requested early reimplantation of a permanent system as the pain relief during the trial had been so complete. This was achieved 3 weeks later, with anchors attached to the deep fascia on the lateral and medial aspect of the knee, just posterior to the joint itself, and the leads tunneled to her anteromedial thigh where a subcutaneous pocket was created at her request for the battery. She achieved total relief once more with the reinstitution of stimulation. On several occasions, it has appeared as though the system has failed, but on each occasion, the patient has realized that the battery had not been charged and relief of pain has returned within 30 minutes after recharging. The system has also required several revisions to date: Lead migration on three separate occasions has been an issue in the context of an elite athlete undergoing highperformance training, the battery has required resiting (tunneled superficial to the inguinal ligament, across the

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Figure 1 Neuromodulation leads placed in subcutaneous tissues of below knee amputation stump. The lateral side of the stump is on the left side of the figure. abdominal wall and between the breasts to a new subcutaneous pocket in the right subclavicular region. An extension was added from a position in the right lower quadrant of the abdominal wall) to facilitate training, and the battery required replacement after 4 months when it unexpectedly failed. She has had problems with falls: She was right footed prior to her amputation, and with the relief of pain, she has had a tendency to forget and try to step out on her right foot. She also describes ‘losing her foot’ with the relief of pain. Prior to the implantation procedure, she described her foot as being twisted backwards and split in two. With onset of the stimulation, her foot reoriented to a normal position, became one again, and then disappeared. She found this latter experience disconcerting. At 6 months, she remains free of pain and on no medication. She feels the neuromodulation stimulus as a ‘tapping’ sensation in her stump. She has been able to mobilize with a prosthesis although there are ongoing issues related to the shape of the end of her tibia and the fit of the prosthesis. Discussion We have been unable to find any previously published descriptions of neuromodulation leads being implanted in the stump of an amputee for the control of PLP. The rationale used for this approach was based on an observation by one of the authors (PBC) that PLP stopped

Peripheral Peripheral Neuromodulation Neuromodulation for for Phantom Phantom Limb Limb Pain Pain for the duration of a peripheral nerve block, a phenomenon which others have also noted [29]. This suggested that peripheral factors played an important role in the phenomenon of PLP. The challenge of that experience was to give the peripheral strategy some degree of permanence. Recent literature has suggested that a week of continuous peripheral nerve blockade may achieve that objective [29] but that had not been our experience. Given that PLP develops from the point at which the limb is amputated, the stump seemed a logical place to place the neuromodulation leads. The patient’s habit of reverting to right footedness with relief of pain suggests a return to her original two-footed cortical map, perhaps via the process of brain readaptation as suggested by Acharya and colleagues [35]. The ‘automatic’ process by which the patient described reverting to this habit and her subsequent need to learn to balance without the input from pain infer that neuroplasticity is bidirectional [36] and adds to the growing body of literature documenting the reversal of pain-related brain changes once pain is relieved [37]. Tracking of the patient’s psychological state indicated fluctuations in mood and affect over the initial 6-month period. A number of significant life events independent of the procedure occurred during this time precipitating spikes in psychosocial stress and emotionality. From a biopsychosocial perspective, it would be expected that such fluctuations would impact on the patient’s subjective experience of pain and/or pain relief. Surprisingly, this was not the case, and she reported consistent and complete pain relief—varying only in response to battery functioning. Psychological assessment of the patient’s preexisting personality structure was undertaken to determine any remarkable trait features explaining these results. Personality profiling revealed a highly driven and achievementfocused individual with a tendency toward perfectionism. Of note, trait neuroticism as measured on the Revised NEO Personality Inventory fell within the average range. The surprising feature for us has been the completeness of the pain relief. The longevity of the effect, as well as the consistency of her reports of the effectiveness of the stimulation despite variation in psychosocial and emotional stressors, gives us some confidence that this is not a placebo phenomenon. We have been encouraged to further explore this treatment option in a structured fashion, including neuroimaging, aware of the implications it has for our understanding of PLP. Acknowledgment They wish to acknowledge the contribution of Marleesa Ly to the preparation of the manuscript. References 1 Nikolajsen L, Jensen TS. Phantom limb pain. Br J Anaesth 2001;87(1):107–16.

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Successful peripheral neuromodulation for phantom limb pain.

For decades, the heterogeneity of the amputee population and the complex interaction of biopsychosocial factors have confounded researchers' attempts ...
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