J Brachial Plex Peripher Nerve Inj 2011; 06(01): e40-e43
DOI: 10.1186/1749-7221-6-6
Case report
Irwin and Schwartzman; licensee BioMed Central Ltd.

Complex regional pain syndrome with associated chest wall dystonia: a case report[*]

David J Irwin
1   Drexel University College of Medicine, Department of Neurology, Philadelphia, PA, USA
,
Robert J Schwartzman
1   Drexel University College of Medicine, Department of Neurology, Philadelphia, PA, USA
› Author Affiliations

Subject Editor:
Further Information

Publication History

01 March 2011

26 September 2011

Publication Date:
23 September 2014 (online)

Abstract

Patients with complex regional pain syndrome (CRPS) often suffer from an array of associated movement disorders, including dystonia of an affected limb. We present a case of a patient with long standing CRPS after a brachial plexus injury, who after displaying several features of the movement disorder previously, developed painful dystonia of chest wall musculature. Detailed neurologic examination found palpable sustained contractions of the pectoral and intercostal muscles in addition to surface allodynia. Needle electromyography of the intercostal and paraspinal muscles supported the diagnosis of dystonia. In addition, pulmonary function testing showed both restrictive and obstructive features in the absence of a clear cardiopulmonary etiology. Treatment was initiated with intrathecal baclofen and the patient had symptomatic relief and improvement of dystonia. This case illustrates a novel form of the movement disorder associated with CRPS with response to intrathecal baclofen treatment.

*This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.


 
  • References

  • 1 Schwartzman RJ, Patel M, Grothusen JR, Alexander GM. Efficacy of 5-day continuous lidocaine infusion for the treatment of refractory complex regional pain syndrome. Pain Med 2009; 10 (2) 401-412 10.1111/j.1526-4637.2009.00573.x 19284488
  • 2 Harden RN, Bruehl S. Diagnostic criteria: the statistical derivation of the four criterion factors. In: Wilson PR, Stanton-Hicks MD, Harden RN. CRPS: Current Diagnosis and Therapy. IASP Press; Seattle: 2005: 45-58
  • 3 Schwartzman RJ, Kerrigan J. The movement disorder of reflex sympathetic dystrophy. Neurology 1990; 40 (1) 57-61 2296383
  • 4 Van Hilten JJ, Blumberg HPD, Schwartzman RJ. Factor IV: Movement disorders and dystrophy: clinical and pathophysiological aspects. In: Wilson PR, Stanton-Hicks MD, Harden RN. CRPS: Current Diagnosis and Therapy. IASP Press; Seattle: 2005: 45-58
  • 5 Schott GD. Peripherally-triggered CRPS and dystonia. Pain 2007; 130: 203-207 10.1016/j.pain.2007.04.013 17512665
  • 6 van Rijn MA, Marinus J, Putter H, van Hilten JJ. Onset and progression of dystonia in complex regional pain syndrome. Pain 2007; 130 (3) 287-293 10.1016/j.pain.2007.03.027 17499924
  • 7 van Hilten JJ, van de Beek WJ, Roep BO. Multifocal or generalized dystonia in complex regional pain syndrome: a distinct clinical entity associated with HLA-DR 13. Ann Neurol 2000; 48: 113-116 10.1002/1531-8249(200007)48:1<113::AID-ANA18>3.0.CO;2-9 10894225
  • 8 van Hilten JJ, van de Beek WJ, Vein AA, van Dijk JG, Middelkoop HA. Clinical aspects of multifocal or generalized tonic dystonia in reflex sympathetic dystrophy. Neurol 2001; 56: 1762-1765
  • 9 Harden RN, Bruehl S, Stanton-Hicks M, Wilson PR. Proposed new diagnostic criteria for complex regional pain syndrome. Pain med 2007; 8: 326-331 10.1111/j.1526-4637.2006.00169.x 17610454
  • 10 Rasmussen J, Grothusen JR, Rosso AL, Schwartzman RJ. Atypical chest pain: evidence of intercostobrachial nerve sensitization in complex regional pain syndrome. Pain Phys 2009; 12: e329-e324
  • 11 Standardization of spirometry- 1987 update. Official statement of the American Thoracic Society. Respir Care 1987; 32 (11) 1039-1060 10315742
  • 12 van de Beek WJ, Vein A, Hilgevoord AA, van Dijk JG, van Hilten BJ. Neurophysiologic aspects of patients with generalized or multifocal tonic dystonia of reflex sympathetic dystrophy. J Clin Neurophysiol 2002; 19 (1) 77-83 10.1097/00004691-200201000-00011 11896357
  • 13 Verdugo R, Ochoa JL. Abnormal movements in complex regional pain syndrome: assessment of their nature. Muscle Nerve 2000; 23: 198-205 10.1002/(SICI)1097-4598(200002)23:2<198::AID-MUS9>3.0.CO;2-4 10639611
  • 14 Braun N, Abd A, Baer J, Blitzer A, Stewart C, Brin M. Dyspnea in dystonia: a functional evaluation. Chest 1995; 107 (5) 1309-1316 10.1378/chest.107.5.1309 7750324
  • 15 Mink JW. Abnormal circuit function in dystonia. Neurol 2006; 66 (7) 959
  • 16 Huang YZ, Trender-Gerhard I, Edwards MJ et al: Motor system inhibition in dopa-responsive dystonia and its modulation by treatment. Neurol 2006; 66 (7) 1088-1090 10.1212/01.wnl.0000214304.03105.f4
  • 17 Tisch S, Limousin P, Rothwell JC et al: Changes in forearm reciprocal inhibition following pallidal stimulation for dystonia. Neurol 2006; 66 (7) 1091-1093 10.1212/01.wnl.0000204649.36458.8f
  • 18 van de Beek WJ, Vein A, Hilgevoord AJ, van Dijk G, van Hilten B. Neurophysiologic aspects of patients with generalized or multifocal tonic dystonia of reflex sympathetic dystrophy. J Clin Neurophysiol 2002; 19 (1) 77-83 10.1097/00004691-200201000-00011 11896357
  • 19 Schouten AC, Van de Beek WJ, Van Hilten JJ, Van der Helm FC. Proprioceptive reflexes in patients with reflex sympathetic dystrophy. Exp Brain Res 2003; 151 (1) 1-8 10.1007/s00221-003-1420-x 12743675
  • 20 Schwenkreis P, Janssen F, Rommel O et al: Bilateral motor cortex disinhibition in complex regional pain syndrome (CRPS) type I of the hand. Neurol 2003; 61 (4) 515-519
  • 21 Weber M, Birklein F, Neundorfer B, Schmelz M. Facilitated neurogenic inflammation in complex regional pain syndrome. Pain 2001; 91 (3) 251-257 10.1016/S0304-3959(00)00445-0 11275381
  • 22 Schouenborg J. Learning in sensorimotor circuits. Curr Opin Neurobiol 2004; 14 (6) 693-697 10.1016/j.conb.2004.10.009 15582370
  • 23 Alexander GM, van Rijn MA, van Hilten JJ, Perreault MJ, Schwartzman RJ. Changes in cerebrospinal fluid levels of pro-inflammatory cytokines in CRPS. Pain 2005; 116: 213-219 10.1016/j.pain.2005.04.013 15964681
  • 24 Alexander GM, Perreault MJ, Reichenberger ER, Schwartzman RJ. Changes in immune and glial markers in the CSF of patients with complex regional pain syndrome. Brain Behav. Immun 2007; 21: 668-676 10.1016/j.bbi.2006.10.009 17129705
  • 25 Del Valle L, Schwartzman RJ, Alexander G. Spinal cord histopathological alterations in a patient with complex regional pain syndrome. Brain Behav. Immunity 2009; 23: 85-91 10.1016/j.bbi.2008.08.004
  • 26 van Rijn MA, Munts AG, Marinus J, Voormolen JHC, de Boer KS, Teepe-Twiss IM, van Dasselaar NT, Delhaas EM, van Hilten JJ. Intrathecal baclofen for dystonia of complex regional pain syndrome. Pain 2009; 143: 41-47 10.1016/j.pain.2009.01.014 19232828
  • 27 Malcangio M, Bowery NG. GABA and its receptors in the spinal cord. Trends Pharmacol Sci 1996; 17: 457-462 10.1016/S0165-6147(96)01013-9 9014500