Accessible Requires Authentication Published by De Gruyter April 1, 2017

Pain reduction due to novel sensory-motor training in Complex Regional Pain Syndrome I – A pilot study

Anne-Christine Schmid, Anja Schwarz, Sylvia M. Gustin, Joel D. Greenspan, Friedhelm C. Hummel and Niels Birbaumer

Abstract

Background and aims

Patients suffering from Complex Regional Pain Syndrome (CRPS) of the upper limb show a changed cortical representation of the affected hand. The lip area invades the former hand area contralateral to the affected hand. This change in cortical representation is correlated to the intensity of ongoing pain in patients with CRPS. Further studies revealed that restoration of the original representation coincides with a decrease of pain. Sensory-motor training protocols can increase and/or relocate cortical somatosensory and motor representation areas of the fingers, as shown, for example, in Braille reading individuals and professional violin players. Further, there is evidence that sensory-motor discrimination training has a beneficial effect on both the intensity of pain and the mislocalization of sensory-motor cortical areas in CRPS patients. Based on these propositions, we developed a novel sensory-motor self-training paradigm for CRPS patients to use in a home-based manner.

Methods

Ten CRPS patients performed the sensory-motor training for 2 weeks. The training consists of a braille-like haptic task with different training modes (bi-manual, speed and memory training). During the training, as well as 1 week before and after, patients were asked to fill out pain diaries. Furthermore, measures of impairment were acquired at baseline and post training.

Results

Patients showed significant pain reduction after the 2 week training period. The overall disability as well as the depression scores showed a trend to improve after the 2 week training. The reduction in pain was correlated with the total amount of training performed.

Conclusions

This is a first proof of principle study of a novel sensory-motor self-training protocol to reduce pain in CRPS patients. The more consistent the patients trained the larger the pain reduction. Sensory-motor training, which can be performed on a regular basis at home might provide a novel interventional strategy to improve symptoms of CRPS.

Implications

Although a larger study needs to be conducted to confirm our findings, including long-term follow-up, the results show, that a sensory-motor home-based training is a strategy worth exploring further for the reduction of pain as well as high frequency training for patients with CRPS.


EPFL SV BMI UPHUMMEL Clinique Romande de Réadaptation (CRR) E1 B2 Campus SUVA Av. Grand-Champsec 90 CP 352 1951 Sion, Switzerland;Brain Mind Institute, SV Centre of Neuroprosthetics (CNP), Swiss Federal Institute of Technology (EPFL), Campus Biotech 9, Chemin des Mines 1202 Geneva, Swizerland.Tel.: +49 40 7410 55573; fax: +49 40 7410 57391.

  1. Author contributions: All authors discussed the results and commented on themanuscript.

  2. Ethical issues: Written informed consent was obtained from all subjects according to the Declaration of Helsinki (www.wma.net/en/30publications) and withapproval from the local Ethics Committee of the Medical Faculty of the University of Tuebingen.

  3. Funding: This study was supported by Bundesministerium für Bildung und Forschung (BMBF grant D 20.01667) and Deutsche Forschungsgesellschaft (DFG grant BI195/63–1).

  4. Conflict of interest: The authors certify that they have NO affiliations with or involvement in any organization or entity with any financial interest (such as honoraria; educational grants; participation in speakers’ bureaus; membership, employment, consultancies, stock ownership, or other equity interest; and expert testimony or patent-licensing arrangements), or non-financial interest (such as personal or professional relationships, affiliations, knowledge or beliefs) in the subject matter or materials discussed in this manuscript.

Acknowledgments

Abd Al-Jabbar Al-Shukry, Merlin Evers, Hendrik Heinzl, Harrison Norman, Daniela Palme, Myriam Wurst helped with the data acquisition.

References

[1] van Velzen GA, Perez RS, van Gestel MA, Huygen FJ, van Kleef M, van Eijs F, Dahan A, van Hilten JJ, Marinus J. Health-related quality of life in 975 patients with complex regional pain syndrome type 1. Pain 2014;155:629–34. Search in Google Scholar

[2] Veldman PH, Reynen HM, Arntz IE, Goris RJ. Signs and symptoms of reflex sympathetic dystrophy: prospective study of 829 patients. Lancet 1993;342:1012–6. Search in Google Scholar

[3] Bruehl S. An update on the pathophysiology of complex regional pain syndrome. Anesthesiology 2010;113:713–25. Search in Google Scholar

[4] Moseley GL. Distorted body image in complex regional pain syndrome. Neurology 2005;65:773. Search in Google Scholar

[5] Moseley GL, Zalucki NM, Wiech K. Tactile discrimination, but not tactile stimulation alone, reduces chronic limb pain. Pain 2008;137:600–8. Search in Google Scholar

[6] O’Connell NE, Wand BM, McAuley J, Marston L, Moseley GL. Interventions for treating pain and disability in adults with complex regional pain syndrome. Cochrane Database Syst Rev 2013;30. Search in Google Scholar

[7] Schmid AC. Pathophysiological aspects of Complex Regional Pain Syndrome (CRPS). Aktuelle Rheumatologie 2011;36:23–7. Search in Google Scholar

[8] Di Pietro F, McAuley JH, Parkitny L, Lotze M, Wand BM, Moseley GL, Stanton TR. Primary somatosensory cortex function in complex regional pain syndrome: a systematic review and meta-analysis. J Pain 2013;14:1001–18. Search in Google Scholar

[9] Haag LM, Heba S, Lenz M, Glaubitz B, Hoffken O, Kalisch T, Puts NA, Edden RA, Tegenthoff M, Dinse H, Schmidt-Wilcke T. Resting BOLD fluctuations in the primary somatosensory cortex correlate with tactile acuity. Cortex 2015;64:20–8. Search in Google Scholar

[10] Classen J, Liepert J, Wise SP, Hallett M, Cohen LG. Rapid plasticity of human cortical movement representation induced by practice. J Neurophysiol 1998;79:1117–23. Search in Google Scholar

[11] Juottonen K, Gockel M, Silen T, Hurri H, Hari R, Forss N. Altered central sensorimotor processing in patients with complex regional pain syndrome. Pain 2002;98:315–23. Search in Google Scholar

[12] Maihofner C, Handwerker HO, Neundorfer B, Birklein F. Patterns of cortical reorganization in complex regional pain syndrome. Neurology 2003;61:1707–15. Search in Google Scholar

[13] Vartiainen N, Kirveskari E, Kallio-Laine K, Kalso E, Forss N. Cortical reorganization in primary somatosensory cortex in patients with unilateral chronic pain. J Pain 2009;10:854–9. Search in Google Scholar

[14] Flor H, Elbert T, Knecht S, Wienbruch C, Pantev C, Birbaumer N, Larbig W, Taub E. Phantom-limb pain as a perceptual correlate of cortical reorganization following arm amputation. Nature 1995;375:482–4. Search in Google Scholar

[15] Pleger B, Tegenthoff M, Schwenkreis P, Janssen F, Ragert P, Dinse HR, Volker B, Zenz M, Maier C. Mean sustained pain levels are linked to hemispherical side-to-side differences of primary somatosensory cortex in the complex regional pain syndrome I. Exp Brain Res 2004;155:115–9. Search in Google Scholar

[16] Sinis N, Birbaumer N, Schwarz A, Gustin S, Unertl K, Schaller HE, Haerle M. [Memantine and Complex Regional Pain Syndrome (CRPS): effects of treatment and cortical reorganisation]. Handchirurgie, Mikrochirurgie, plastische Chirurgie: Organ der Deutschsprachigen Arbeitsgemeinschaft fur Handchirurgie: Organ der Deutschsprachigen Arbeitsgemeinschaft fur Mikrochirurgie der Peripheren Nerven und Gefasse: Organ der Vereinigung der Deutschen Plastischen Chirurgen 2006;38:164–71. Search in Google Scholar

[17] Elbert T, Pantev C, Wienbruch C, Rockstroh B, Taub E. Increased cortical representation of the fingers of the left hand in string players. Science 1995;270:305–7. Search in Google Scholar

[18] Harris JA, Harris IM, Diamond ME. The topography of tactile learning in humans. J Neurosci 2001;21:1056–61. Search in Google Scholar

[19] Pascual-Leone A, Torres F. Plasticity of the sensorimotor cortex representation of the reading finger in Braille readers. Brain 1993;116:39–52. Search in Google Scholar

[20] Moseley GL, Wiech K. The effect of tactile discrimination training is enhanced when patients watch the reflected image of their unaffected limb during training. Pain 2009;144:314–9. Search in Google Scholar

[21] Lewis JS, Schweinhardt P. Perceptions of the painful body: the relationship between body perception disturbance, pain and tactile discrimination in complex regional pain syndrome. Eur J Pain 2012;16:1320–30. Search in Google Scholar

[22] Peltz E, Seifert F, Lanz S, Muller R, Maihofner C. Impaired hand size estimation in CRPS. J Pain 2011. Search in Google Scholar

[23] Harden RN, Bruehl S, Perez RS, Birklein F, Marinus J, Maihofner C, Lubenow T, Buvanendran A, Mackey S, Graciosa J, Mogilevski M, Ramsden C, Chont M, Vatine JJ. Validation of proposed diagnostic criteria (the “Budapest Criteria”) for Complex Regional Pain Syndrome. Pain 2010;150:268–74. Search in Google Scholar

[24] Harden RN, Bruehl SP. Diagnosis of complex regional pain syndrome: signs, symptoms, and new empirically derived diagnostic criteria. Clin J Pain 2006;22:415–9. Search in Google Scholar

[25] Oldfield RC. The assessment and analysis of handedness: the Edinburgh inventory. Neuropsychologia 1971;9:97–113. Search in Google Scholar

[26] Rolke R, Baron R, Maier C, Tolle TR, Treede RD, Beyer A, Binder A, Birbaumer N, Birklein F, Botefur IC, Braune S, Flor H, Huge V, Klug R, Landwehrmeyer GB, Magerl W, Maihofner C, Rolko C, Schaub C, Scherens A, Sprenger T, Valet M, Wasserka B. Quantitative sensory testing in the German Research Network on Neuropathic Pain (DFNS): standardized protocol and reference values. Pain 2006;123:231–43. Search in Google Scholar

[27] Rolke R, Magerl W, Campbell KA, Schalber C, Caspari S, Birklein F, Treede RD. Quantitative sensory testing: a comprehensive protocol for clinical trials. Eur J Pain 2006;10:77–88. Search in Google Scholar

[28] Perez MA, Cohen LG. Mechanisms underlying functional changes in the primary motor cortex ipsilateral to an active hand. J Neurosci 2008;28: 5631–40. Search in Google Scholar

[29] Perez MA, Tanaka S, Wise SP, Sadato N, Tanabe HC, Willingham DT, Cohen LG. Neural substrates of intermanual transfer of a newly acquired motor skill. Curr Biol: CB 2007;17:1896–902. Search in Google Scholar

[30] Tait RC, Pollard CA, Margolis RB, Duckro PN, Krause SJ. The Pain Disability Index: psychometric and validity data. Arch Phys Med Rehabil 1987;68:438–41. Search in Google Scholar

[31] Hautzinger M. Die CES-D-Skala. ein Depressionsmessinstrument für Untersucungen in der Allgemeinbevölkerung. Diagnostica 1988;34:167–73. Search in Google Scholar

[32] Braun C, Haug M, Wiech K, Birbaumer N, Elbert T, Roberts LE. Functional organization of primary somatosensory cortex depends on the focus of attention. Neuroimage 2002;17:1451–8. Search in Google Scholar

[33] Kwakkel G, van Peppen R, Wagenaar RC, Wood Dauphinee S, Richards C, Ashburn A, Miller K, Lincoln N, Partridge C, Wellwood I, Langhorne P. Effects of augmented exercise therapy time after stroke: a meta-analysis. Stroke 2004;35:2529–39. Search in Google Scholar

[34] Maihofner C, Handwerker HO, Neundorfer B, Birklein F. Cortical reorganization during recovery from complex regional pain syndrome. Neurology 2004;63:693–701. Search in Google Scholar

[35] Gustin SM, Schwarz A, Birbaumer N, Sines N, Schmid AC, Veit R, Larbig W, Flor H, Lotze M. NMDA-receptor antagonist and morphine decrease CRPS-pain and cerebral pain representation. Pain 2010;151:69–76. Search in Google Scholar

[36] Moseley GL. Is successful rehabilitation of complex regional pain syndrome due to sustained attention to the affected limb? A randomised clinical trial. Pain 2005;114:54–61. Search in Google Scholar

[37] Hummel FC, Cohen LG. Drivers of brain plasticity. Curr Opin Neurol 2005;18:667–74. Search in Google Scholar

[38] Reis J, Schambra HM, Cohen LG, Buch ER, Fritsch B, Zarahn E, Celnik PA, Krakauer JW. Noninvasive cortical stimulation enhances motor skill acquisition over multiple days through an effect on consolidation. Proc Natl Acad Sci USA 2009;106:1590–5. Search in Google Scholar

[39] Zimerman M, Nitsch M, Giraux P, Gerloff C, Cohen LG, Hummel FC. Neuroenhancement of the aging brain: restoring skill acquisition in old subjects. Ann Neurol 2013;73:10–5. Search in Google Scholar

[40] Zimerman M, Heise KF, Hoppe J, Cohen LG, Gerloff C, Hummel FC. Modulation of training by single-session transcranial direct current stimulation to the intact motor cortex enhances motor skill acquisition of the paretic hand. Stroke 2012;43:2185–91. Search in Google Scholar

[41] Wessel MJ, Zimerman M, Hummel FC. Non-invasive brain stimulation: an interventional tool for enhancing behavioral training after stroke. Front Hum Neurosci 2015;9:265. Search in Google Scholar

[42] Kasschau M, Reisner J, Sherman K, Bikson M, Datta A, Charvet LE. Transcranial direct current stimulation is feasible for remotely supervised home delivery in multiple sclerosis. Neuromodulation 2016. Search in Google Scholar

Abbreviations

CES-D

Center for Epidemiological Studies – Depression Scale

CRPS

Complex Regional Pain Syndrome

M1

Primary motor cortex

PDI

Pain DisabilityIndex

PT

Physical Therapy

S1

Primary sensory cortex

ST

Sensory-motor Training

tDCS

Transcranial Direct Current Stimulation

TMS

Transcranial Magnetic Stimulation

VAS

Visual Analogue Scale.

Appendix A. Supplementary data

Supplementary data associated with this article can be found, in the online version, at http://dx.doi.Org/10.1016/j.sjpain.2016.11.003.

Received: 2016-07-04
Revised: 2016-11-03
Accepted: 2016-11-09
Published Online: 2017-04-01
Published in Print: 2017-04-01

© 2016 Scandinavian Association for the Study of Pain