Restoration of erect posture in idiopathic camptocormia by electrical stimulation of the globus pallidus internus

Report of 2 cases

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  • 1 Unit of Functional and Stereotactic Neurosurgery, Department of Neurosurgery, University of Athens Medical School, Evangelismos Hospital; and
  • 2 Department of Psychiatry, University of Athens Medical School, Eginition Hospital, Athens, Greece
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The authors report on 2 young patients who developed drug-resistant idiopathic dystonic camptocormia (bent spine) and were treated successfully by deep brain stimulation (DBS) of the globus pallidus internus (GPi). The first patient, a 26-year-old woman, suffered for 3 years from such severe camptocormia that she became unable to walk and was confined to bed or a wheelchair. The second patient, a 21-year-old man, suffered for 6 months from less severe camptocormia; he was able to walk but only for short distances with a very bent spine, the arms in a parallel position to the legs, and the hands almost approaching the floor to potentially support him in case of a forward fall. Within a few days following DBS, both patients experienced marked clinical improvement. At most recent follow-up (44 months in one case and 42 in the other), the patients' ability to walk upright remained normal. Similar findings have only been reported recently in a few cases of camptocormia secondary to Parkinson disease or tardive dyskinesia. On the basis of the experience of these 2 idiopathic cases and the previously reported cases of secondary camptocormia with a favorable response to GPi DBS, the authors postulate that specific patterns of oscillatory activity in the GPi are vital for the maintenance of erect posture and the adoption of bipedal walking by humans.

Abbreviations used in this paper: DBS = deep brain stimulation; DSM-IV-TR = Diagnostic and Statistical Manual of Mental Disorders, fourth edition, text revision; GPi = globus pallidus internus; PD = Parkinson disease.

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Contributor Notes

Address correspondence to: Damianos E. Sakas, M.D., Department of Neurosurgery, University of Athens Medical School, Evangelismos Hospital, 4 Marasli Street, 10676 Athens, Greece. email: sakasde@med.uoa.gr.

Please include this information when citing this paper: published online April 9, 2010; DOI: 10.3171/2010.3.JNS09981.

  • 1

    Albin RL, , Young AB, & Penney JB: The functional anatomy of basal ganglia disorders. Trends Neurosci 12:366375, 1989

  • 2

    American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders, ed 4. Text Revision: DSM-IV-TRTM Washington, DC, American Psychiatric Association, 2000

    • Search Google Scholar
    • Export Citation
  • 3

    Azher SN, & Jankovic J: Camptocormia: pathogenesis, classification, and response to therapy. Neurology 65:355359, 2005

  • 4

    Bar-Gad I, , Morris G, & Bergman H: Information processing, dimensionality reduction and reinforcement learning in the basal ganglia. Prog Neurobiol 71:439473, 2003

    • Search Google Scholar
    • Export Citation
  • 5

    Brown P, & Eusebio A: Paradoxes of functional neurosurgery: clues from basal ganglia recordings. Mov Disord 23:1220, 2008

  • 6

    Crompton RH, , Vereecke EE, & Thorpe SK: Locomotion and posture from the common hominoid ancestor to fully modern hominins, with special reference to the last common panin/hominin ancestor. J Anat 212:501543, 2008

    • Search Google Scholar
    • Export Citation
  • 7

    DeLong MR: Primate models of movement disorders of basal ganglia origin. Trends Neurosci 13:281285, 1990

  • 8

    Djaldetti R, & Melamed E: Camptocormia in Parkinson's disease: new insights. J Neurol Neurosurg Psychiatry 77:1205, 2006

  • 9

    Fahn S, & Williams DT: Psychogenic dystonia. Adv Neurol 50:431455, 1988

  • 10

    Ferraye MU, , Debû B, , Fraix V, , Goetz L, , Ardouin C, & Yelnik J, : Effects of pedunculopontine nucleus area stimulation on gait disorders in Parkinson's disease. Brain 133:205214, 2010

    • Search Google Scholar
    • Export Citation
  • 11

    Geyer HL, & Bressman SB: The diagnosis of dystonia. Lancet Neurol 5:780790, 2006

  • 12

    Inzelberg R, , Hattori N, , Nisipeanu P, , Abo Mouch S, , Blumen SC, & Carasso RL, : Camptocormia, axial dystonia, and parkinsonism: phenotypic heterogeneity of a parkin mutation. Neurology 60:13931394, 2003

    • Search Google Scholar
    • Export Citation
  • 13

    Jahn K, , Deutschländer A, , Stephan T, , Kalla R, , Wiesmann M, & Strupp M, : Imaging human supraspinal locomotor centers in brainstem and cerebellum. Neuroimage 39:786792, 2008

    • Search Google Scholar
    • Export Citation
  • 14

    Karbowski K: The old and the new camptocormia. Spine 24:14941498, 1999

  • 15

    Krauss JK, , Yianni J, , Loher TJ, & Aziz TZ: Deep brain stimulation for dystonia. J Clin Neurophysiol 21:1830, 2004

  • 16

    Laroche M, , Delisle MB, , Aziza R, , Lagarrigue J, & Mazieres B: Is camptocormia a primary muscular disease?. Spine 20:10111016, 1995

  • 17

    Marsden CD, & Obeso JA: The functions of the basal ganglia and the paradox of stereotaxic surgery in Parkinson's disease. Brain 117:877897, 1994

    • Search Google Scholar
    • Export Citation
  • 18

    Michelli F, , Cersósimo M, & Piedimonte F: Camptocormia in a patient with Parkinson disease: beneficial effects of pallidal deep brain stimulation. Case report. J Neurosurg 103:10811083, 2005

    • Search Google Scholar
    • Export Citation
  • 19

    Nandi D, , Parkin S, , Scott R, , Winter JL, , Joint C, & Gregory R, : Camptocormia treated with bilateral pallidal stimulation. J Neurosurg 97:461466, 2002

    • Search Google Scholar
    • Export Citation
  • 20

    Nieves AV, , Miyasaki JM, & Lang AE: Acute onset dystonic camptocormia caused by lenticular lesions. Mov Disord 16:177180, 2001

  • 21

    Sakas DE, , Kouyialis AT, , Boviatsis EJ, , Panourias IG, , Stathis P, & Tagaris G: Technical aspects and considerations of deep brain stimulation surgery for movement disorders. Acta Neurochir Suppl 97:(Pt 2) 163170, 2007

    • Search Google Scholar
    • Export Citation
  • 22

    Sakas DE, , Panourias IG, , Boviatsis EJ, , Themistocleous MS, , Stavrinou LC, & Stathis P, : Treatment of idiopathic head drop (camptocephalia) by deep brain stimulation of the globus pallidus internus. Case report. J Neurosurg 110:12711273, 2009

    • Search Google Scholar
    • Export Citation
  • 23

    Sako W, , Nishio M, , Maruo T, , Shimazu H, , Matsuzaki K, & Tamura T, : Subthalamic nucleus deep brain stimulation for camptocormia associated with Parkinson's disease. Mov Disord 15:10761079, 2009

    • Search Google Scholar
    • Export Citation
  • 24

    Schäbitz WR, , Glatz K, , Schuhan C, , Sommer C, , Berger C, & Schwaninger M, : Severe forward flexion of the trunk in Parkinson's disease: focal myopathy of the paraspinal muscles mimicking camptocormia. Mov Disord 18:408414, 2003

    • Search Google Scholar
    • Export Citation
  • 25

    Sławek J, , Derejko M, & Lass P: Camptocormia as a form of dystonia in Parkinson's disease. Eur J Neurol 10:107108, 2003

  • 26

    Souques A, & Rosanoff-Saloff A: La camptocormie. Rev Neurol 22:937939, 1915

  • 27

    Tarsy D, & Simon DK: Dystonia. N Engl J Med 355:818829, 2006

  • 28

    Umapathi T, , Chaudhry V, , Cornblath D, , Drachman D, , Griffin J, & Kuncl R: Head drop and camptocormia. J Neurol Neurosurg Psychiatry 73:17, 2002

    • Search Google Scholar
    • Export Citation
  • 29

    Vitek JL: Pathophysiology of dystonia: a neuronal model. Mov Disord 17:3 Suppl 3 S49S62, 2002

  • 30

    Wunderlich S, , Csoti I, , Reiners K, , Günthner-Lengsfeld T, , Schneider C, & Becker G, : Camptocormia in Parkinson's disease mimicked by focal myositis of the paraspinal muscles. Mov Disord 17:598600, 2002

    • Search Google Scholar
    • Export Citation

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