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Case report
Impact of the use of neurostimulation for the management of a patient with complex pain syndrome and severe trophic changes. Case report
Impacto del manejo con neuroestimulación en un paciente con síndrome doloroso complejo y cambios tróficos severos. Reporte de caso
Mario H. Villegas Pinedaa, Christian Herrerab, Tatiana León Martínezc, Vasco Ordoñez Fernándezc,
Corresponding author
vasof82@hotmail.com

Corresponding author at: Anaesthesia Department, Hospital Universitario del Valle Medical School – Calle 5 # 36-08 piso 4, Cali, Colombia.
a Anaesthetist and Pain Specialist, Pain Clinic Coordinator, Fundación Valle de Lili, Cali, Colombia
b Anaesthetist and Pain Specialist, Pain Clinic, Fundación Valle de Lili, Cali, Colombia
c Resident 2. año, Department of Anesthesiology, Universidad del Valle, Cali, Colombia
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bone fractures being the most frequently associated with it&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The pathophysiology of CRPS is still not fully understood&#46; There are multiple mechanisms that play an important role in its onset and maintenance&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Several treatments have been proposed for pain relief&#44; functional recovery and psychological improvement&#44; but management is often difficult and the functionality of the affected limb may be compromised if not treated early on&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Patient information</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 55-year-old male patient&#44; street vendor&#44; with no important medical history&#44; who fell from a 1-m height and sustained trauma to the right hand 8 years before&#46; He then developed oedema and pain in the limb&#46; When seen by the orthopaedic service&#44; he was diagnosed with a &#8220;sprained wrist&#8221; and was ordered splinting&#44; anti-inflammatory drugs and physical therapy&#44; with no improvement&#46; The patient came back&#44; complaining of persistent pain and progressive oedema that required splint removal&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Clinical findings</span><p id="par0015" class="elsevierStylePara elsevierViewall">The patient reported hyperesthesia and persistent hyperalgesia in dermatomes C5&#8211;C8&#44; 5&#47;10 on the visual analogue scale&#44; increased oedema involving the hand&#44; the forearm and the arm&#44; and functional limitation of the distal portion of the right upper limb&#44; with discoloration of the surrounding skin&#46; These findings led to the diagnosis of CRPS type I and treatment was initiated with amitriptyline 35<span class="elsevierStyleHsp" style=""></span>mg&#47;night&#44; carbamazepine 800<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#44; acetaminophen 1<span class="elsevierStyleHsp" style=""></span>g every 6<span class="elsevierStyleHsp" style=""></span>h&#44; and tramadol 4 capsules&#47;day without significant improvement&#46; The patient was then lost to follow-up&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The patient returned three years later with worsened symptoms after sustaining new trauma&#46; The patient showed nail atrophy&#44; absence of hair &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; diaphoresis in the involved area&#44; and marked allodynia besides the symptoms described above&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0070">Therapeutic intervention</span><p id="par0025" class="elsevierStylePara elsevierViewall">For this relapse&#44; the regimen selected was prednisolone for 10 days&#44; hydrocodone 10<span class="elsevierStyleHsp" style=""></span>mg every 8<span class="elsevierStyleHsp" style=""></span>h&#44; and a cervical sympathetic block &#40;CSB&#41;&#46; Two ipsilateral cervical sympathetic blocks were performed and the patient continued on oral medication&#44; with a 5&#37; subjective improvement&#46; However&#44; the patient recurred after some months and&#44; at this point&#44; it was decided to try neurostimulation and continue the management only with hydrocodone&#44; gabapentin and acetaminophen&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The neurostimulation &#40;NS&#41; trial was performed with a 100&#37; improvement and marked oedema reduction &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#46;&#41;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0075">Results and follow-up</span><p id="par0035" class="elsevierStylePara elsevierViewall">After completing the trial&#44; the patient again reported a pain score of 10&#47;10 and all the symptoms returned&#46; This finding led to the prescription of definitive implantation of the neurostimulator&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">After one month of definitive NS implantation&#44; the patient reported &#8220;100&#37; improvement of pain&#8221;&#46; There was marked oedema reduction and progressive return to the normal appearance of the hand and reduced functional impairment&#46; With physical therapy&#44; the patient was able to take up work slowly after one year and the affected area acquired almost normal characteristics&#44; with only mild residual limitation of finger flexion &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0080">Discussion</span><p id="par0045" class="elsevierStylePara elsevierViewall">Several theories have been proposed to explain the cellular and biochemical events that might give rise to CRPS&#46; These include peripheral mechanisms such as axonal and tissue hypoxia secondary to vasoconstriction associated with a nitric oxide synthesis imbalance and increased endothelin-1 and pro-inflammatory interleukin &#40;IL6&#41; production&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9&#44;10</span></a> The diagnosis of CRPS is based on clinical signs and symptoms found in the initial assessment&#46; Since 1994&#44; the International Association for the Study of Pain &#40;IASP&#41;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> has proposed diagnostic criteria to provide very simple and accurate parameters that guide clinicians in the diagnosis of this disorder&#46; More recently&#44; Harden<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> proposed some diagnostic criteria based specifically on the symptomatology of this complex syndrome&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">CRPS shares treatment and management similarities with neuropathic pain&#46; The usual treatments are tricyclic antidepressants&#44; serotonin and noradrenalin reuptake inhibitors&#44; first and second generation anticonvulsants and&#44; occasionally&#44; muscle relaxants&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13&#44;14</span></a> This was the first line of treatment used in our patient&#44; with a very poor response&#46; Opioids may play a limited role in the management of the intractable pain&#44; which is characteristic of this disorder&#46; Concomitant management with steroids may be effective given their anti-inflammatory effect&#44; particularly during the early phase&#46; Our patient received opioids initially with no improvement&#44; and steroids were given in a later stage&#44; which might explain the therapeutic failure of medication use&#46; Interventional treatments are usually indicated when conventional therapy fails to control pain &#40;persistence of pain &#62;4&#47;10 on the visual analogue scale&#41; and the associated symptoms&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a> It has been suggested that limb immobilization may be associated with the onset of CRPS as happened in this case&#44; since it increases pressure&#44; and that early complaints of compression are predictive factors for CRPS&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">The initial work on electrical stimulation for the management of CRPS was based on the gating theory described by Melzak and Wall&#44;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">15&#44;16</span></a> which suggests that intermittent stimulation of A-alpha and beta fibres localized in the spinal dorsal columns would break the vicious circle of central retransmission from peripheral nociceptors in C fibres&#46; To this date&#44; there is no strong evidence regarding the usefulness of spinal NS for the management of CRPS&#46; Considering that some of the NS procedures are relatively new&#44; there is still little evidence in relation to their use and cost- effectiveness&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">17</span></a> Our patient&#39;s symptoms are consistent with the classical CRPS type 1 presentation where the management with multiple drugs and physical therapy did not lead to significant improvement&#44; prompting the use of CSB with partial improvement&#46; The decision to use a NS in this case was based on the transient success with this intervention followed by relapse&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">17&#44;18</span></a> There are case reports that may support the use of this interventional treatment when medical therapies fail&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Several sympathetic&#44; regional intravenous and epidural blocks may be given on an outpatient basis&#46; However&#44; responses to sympathetic blocks vary and they appear to be more effective than placebo in terms of duration but not so of the degree of pain relief&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">19</span></a> NS has been considered the definitive treatment of CRPS both through spinal stimulation for CRPS type I as well as through peripheral nerve stimulation for CRPS type II&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">20</span></a> Forouzanfar studied the long-term effects of cervical and lumbar NS in patients with CRPS I and found that pain intensity was reduced after six months&#44; one and two years after the implantation&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">21</span></a> Kemler conducted a two-year study to assess the impact of NS on CRPS and found pain reduction and improved quality of life&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">22</span></a> Other favourable outcomes have been observed with the use of NS in CRPS including the absence of hyperpathia&#44; normalized temperature sustainability&#44; improved functionality when combine with physical therapy&#44; and a noticeable reduction in the use of analgesics&#46;<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">23&#44;24</span></a> Our patient showed improvement of all his symptoms after the interventional procedure&#44; with just a mild reduction of finger mobility on flexion and extension&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">In conclusion&#44; we suggest that NS may have a noticeable impact on the course of CRPS in patients with recurrent symptoms despite medical management and even despite the use of classical interventional strategies&#46; The rapid onset and development of associated trophic changes could be an indication to intervene early on because of the functional and psychological repercussions for these patients who are usually in the productive stage of their lives&#46; Our patient improved of his symptoms thanks to his therapeutic management&#44; and was able to return to work&#46; He still attends the pain clinic and gave his consent to the publication of his case&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0085">Funding</span><p id="par0070" class="elsevierStylePara elsevierViewall">None&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0090">Conflicts of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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            1 => "Introducci&#243;n"
            2 => "Descripci&#243;n del caso y resultados"
            3 => "Conclusi&#243;n"
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          "titulo" => "Introduction"
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          "titulo" => "Therapeutic intervention"
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          "titulo" => "Results and follow-up"
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          "titulo" => "Discussion"
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    "fechaRecibido" => "2013-06-23"
    "fechaAceptado" => "2014-04-10"
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          "clase" => "keyword"
          "titulo" => "Keywords"
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          "palabras" => array:3 [
            0 => "Complex regional pain syndrome"
            1 => "Neurostimulation"
            2 => "Causalgia"
          ]
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          "clase" => "keyword"
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          "palabras" => array:3 [
            0 => "S&#237;ndrome doloroso regional complejo"
            1 => "Neuroestimulaci&#243;n"
            2 => "Causalgia"
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      ]
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      "en" => array:2 [
        "titulo" => "Abstract"
        "resumen" => "<span class="elsevierStyleSectionTitle" id="sect0010">Introduction</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Limb trauma and surgery are frequent causes of complex regional pain syndrome &#40;CRPS&#41;&#46; Some cases are very difficult to manage despite the use of high-dose analgesics&#44; anti-inflammatory agents and physical therapy&#59; hence the need to look for interventional therapies to slow its progression&#46;</p> <span class="elsevierStyleSectionTitle" id="sect0015">Case description and results</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We present the case of a patient diagnosed with CRPS type I with severe trophic changes and marked functional limitation&#44; managed with multiple pharmacological therapies and nerve blocks without apparent improvement&#46; The patient decided to try neurostimulation with favourable results and substantial improvement months later reported as &#8220;100&#37; improvement&#8221; of pain&#59; reduced oedema&#59; progressive recovery of the normal appearance of the hand&#59; partial recovery of strength&#44; and improved function attributable to the use of this additional therapy&#46;</p> <span class="elsevierStyleSectionTitle" id="sect0020">Conclusion</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Neurostimulation has a noticeable impact on the course of the complex regional pain syndrome accompanied by trophic changes in patients with a poor response to the recommended pharmacological management&#46; Early initiation of this intervention facilitates functional and psychological recovery of the patients&#44; who are usually in the productive stage of their lives&#46;</p>"
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        "titulo" => "Resumen"
        "resumen" => "<span class="elsevierStyleSectionTitle" id="sect0030">Introducci&#243;n</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Causas frecuentes de s&#237;ndrome doloroso regional complejo &#40;SDRC&#41; son el trauma y la cirug&#237;a en extremidades&#46; Algunos casos resultan de muy dif&#237;cil manejo a pesar de la utilizaci&#243;n de altas dosis de analg&#233;sicos&#44; antiinflamatorios y terapia f&#237;sica lo cual motiva la b&#250;squeda de terapias intervencionistas que frenen su progresi&#243;n&#46;</p> <span class="elsevierStyleSectionTitle" id="sect0035">Descripci&#243;n del caso y resultados</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Presentamos el caso de un paciente a quien se le diagnostic&#243; SDRC tipo i con cambios tr&#243;ficos severos y marcada limitaci&#243;n funcional&#44; que fue manejado con m&#250;ltiples terapias farmacol&#243;gicas y bloqueos nerviosos sin mejor&#237;a evidente&#44; a quien posteriormente se decidi&#243; realizar una prueba con neuroestimulaci&#243;n&#44; la cual fue favorable&#46; Meses despu&#233;s se obtuvo una mejor&#237;a sustancial ya que present&#243; mejor&#237;a del dolor &#171;de un100&#37;&#187;&#59; disminuci&#243;n del edema y retorno progresivo al aspecto normal de la mano&#44; recuperaci&#243;n parcial de la fuerza y disminuci&#243;n de su limitaci&#243;n funcional&#44; explicable por la adici&#243;n de esta terapia de manera definitiva&#46;</p> <span class="elsevierStyleSectionTitle" id="sect0040">Conclusi&#243;n</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">La neuroestimulaci&#243;n ejerce un impacto notable sobre la evoluci&#243;n del SDRC con cambios tr&#243;ficos en pacientes con pobre respuesta al manejo farmacol&#243;gico indicado&#46; El inicio temprano de esta intervenci&#243;n facilita la recuperaci&#243;n funcional y psicol&#243;gica al paciente que&#44; usualmente&#44; se encuentra en una etapa productiva de su vida&#46;</p>"
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    "NotaPie" => array:1 [
      0 => array:2 [
        "etiqueta" => "&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Villegas Pineda MH&#44; Herrera C&#44; Mart&#237;nez TL&#44; Fern&#225;ndez VO&#46; Impacto del manejo con neuroestimulaci&#243;n en un paciente con s&#237;ndrome doloroso complejo y cambios tr&#243;ficos severos&#46; Reporte de caso&#46; Rev Colomb Anestesiol&#46; 2014&#59;42&#58;321&#8211;324&#46;</p>"
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Article information
ISSN: 22562087
Original language: English
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2021 February 14 7 21
2021 January 22 5 27
2020 December 9 7 16
2020 November 12 3 15
2020 October 8 5 13
2020 September 3 6 9
2020 August 5 5 10
2020 July 6 5 11
2020 June 3 4 7
2020 May 2 3 5
2020 April 3 2 5
2020 March 4 2 6
2020 February 2 4 6
2020 January 3 3 6
2019 December 4 5 9
2019 November 0 2 2
2019 October 3 4 7
2019 September 0 1 1
2019 July 1 10 11
2019 June 0 2 2
2019 May 3 13 16
2018 December 1 0 1
2018 September 1 0 1
2018 June 2 3 5
2018 May 26 6 32
2018 April 32 7 39
2018 March 25 14 39
2018 February 23 9 32
2018 January 36 9 45
2017 December 23 8 31
2017 November 29 3 32
2017 October 26 15 41
2017 September 32 9 41
2017 August 50 11 61
2017 July 49 8 57
2017 June 56 10 66
2017 May 54 9 63
2017 April 48 15 63
2017 March 33 6 39
2017 February 21 9 30
2017 January 10 5 15
2016 December 40 18 58
2016 November 44 12 56
2016 October 27 15 42
2016 September 54 7 61
2016 August 45 9 54
2016 July 23 6 29
2016 June 2 9 11
2016 May 2 18 20
2016 March 2 0 2
2016 February 0 17 17
2016 January 0 14 14
2015 December 18 6 24
2015 November 46 11 57
2015 October 55 11 66
2015 September 38 11 49
2015 August 33 5 38
2015 July 38 8 46
2015 June 28 5 33
2015 May 34 9 43
2015 April 23 8 31
2015 March 39 20 59
2015 February 39 12 51
2015 January 43 11 54
2014 December 69 15 84
2014 November 43 9 52
2014 October 55 18 73
2014 September 15 7 22
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos