Camptocormia treated with bilateral pallidal stimulation

Case report

Dipankar Nandi M.B.B.S., M.Chir.1, Simon Parkin M.R.C.P.1, Richard Scott Ph.D.1, Jonathan L. Winter1, Carole Joint R.G.N.1, Ralph Gregory F.R.C.P.1, John Stein F.R.C.P.1, and Tipu Z. Aziz F.R.C.S.1
View More View Less
  • 1 University Laboratory of Physiology, University of Oxford; Departments of Neurology, Neurosurgery, and Neuropsychology, Radcliffe Infirmary, Oxford; and Department of Neurology, Imperial College School of Medicine, London, United Kingdom
Restricted access

Purchase Now

USD  $45.00

JNS + Pediatrics - 1 year subscription bundle (Individuals Only)

USD  $505.00

JNS + Pediatrics + Spine - 1 year subscription bundle (Individuals Only)

USD  $600.00
Print or Print + Online

✓ The authors report the neurological, neurophysiological, and neuropsychological effects of using long-term bilateral pallidal high-frequency deep brain stimulation (DBS) in a case of disabling camptocormia. Deep brain stimulation electrodes were implanted stereotactically to target the globus pallidus internus (GPi) bilaterally. Local field potentials (FPs) were recorded using the DBS electrodes and concurrent abdominal flexor electromyography (EMG) potentials during camptocormic episodes. Videotaped assessments of the movement disorder and neuropsychological evaluations of the patient before implantation and 6 months after initiation of pallidal stimulation were recorded.

There was significant functional improvement following long-term pallidal stimulation, and some improvement was noted in neuropsychological scores. A temporal correlation between the GPi FPs and EMG-recorded rectus abdominis potentials was evident. There were no treatment-related adverse effects. The authors have found that long-term pallidal stimulation was safe and offered functional benefit to a patient with this severely disabling condition. The physiological studies may help further the understanding of the pathophysiology of this rare entity.

JNS + Pediatrics - 1 year subscription bundle (Individuals Only)

USD  $505.00

JNS + Pediatrics + Spine - 1 year subscription bundle (Individuals Only)

USD  $600.00

Contributor Notes

Address reprint requests to: Tipu Z. Aziz, F.R.C.S., Department of Neurosurgery, Radcliffe Infirmary, Woodstock Road, Oxford OX2 6HE, United Kingdom. email: tipu.aziz@physiol.ox.ac.uk.
  • 1.

    American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders: DSM-IV, ed 4. Washington DC: American Psychiatric Association, 1994 American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders: DSM-IV, ed 4. Washington DC: American Psychiatric Association, 1994

    • Search Google Scholar
    • Export Citation
  • 2.

    Ballenger JC: A case of camptocormia occurring in psychotherapy. J Nerv Ment Dis 162:291294, 1976 Ballenger JC: A case of camptocormia occurring in psychotherapy. J Nerv Ment Dis 162:291–294, 1976

    • Search Google Scholar
    • Export Citation
  • 3.

    Benabid AL, , Benazzouz A, & Hoffmann D, et al. : Long-term electrical inhibition of deep brain targets in movement disorders. Mov Disord 13 (Suppl 3):119125, 1998 Benabid AL, Benazzouz A, Hoffmann D, et al: Long-term electrical inhibition of deep brain targets in movement disorders. Mov Disord 13 (Suppl 3):119–125, 1998

    • Search Google Scholar
    • Export Citation
  • 4.

    Bergner M, , Bobbitt RA, & Carter WB, et al. : The Sickness Impact Profile: development and final revision of a health status measure. Med Care 19:787805, 1981 Bergner M, Bobbitt RA, Carter WB, et al: The Sickness Impact Profile: development and final revision of a health status measure. Med Care 19:787–805, 1981

    • Search Google Scholar
    • Export Citation
  • 5.

    Djaldetti R, , Mosberg-Galili R, & Sroka H, et al. : Camptocormia (bent spine) in patients with Parkinson's disease—characterization and possible pathogenesis of an unusual phenomenon. Mov Disord 14:443447, 1999 Djaldetti R, Mosberg-Galili R, Sroka H, et al: Camptocormia (bent spine) in patients with Parkinson's disease—characterization and possible pathogenesis of an unusual phenomenon. Mov Disord 14:443–447, 1999

    • Search Google Scholar
    • Export Citation
  • 6.

    Garcia de Yebenes J, & Garcia Ruiz-Espiga P: Descripcion princeps de la camptocormia en la enfermedad de Parkinson por Francisco de Zurbaran a mediados del siglo XVII. Neurologia 15:265266, 2000 Garcia de Yebenes J, Garcia Ruiz-Espiga P: Descripcion princeps de la camptocormia en la enfermedad de Parkinson por Francisco de Zurbaran a mediados del siglo XVII. Neurologia 15:265–266, 2000

    • Search Google Scholar
    • Export Citation
  • 7.

    Ghika J, , Villemure JG, & Fankhauser H, et al. : Efficiency and safety of bilateral contemporaneous pallidal stimulation (deep brain stimulation) in levodopa-responsive patients with Parkinson's disease with severe motor fluctuations: a 2-year follow-up review. J Neurosurg 89:713718, 1998 Ghika J, Villemure JG, Fankhauser H, et al: Efficiency and safety of bilateral contemporaneous pallidal stimulation (deep brain stimulation) in levodopa-responsive patients with Parkinson's disease with severe motor fluctuations: a 2-year follow-up review. J Neurosurg 89:713–718, 1998

    • Search Google Scholar
    • Export Citation
  • 8.

    Gomez EA, & Drooby AS: Camptocormia in a case of manic-depressive disorder. Psychosomatics 28:592, 594595, 1987 Gomez EA, Drooby AS: Camptocormia in a case of manic-depressive disorder. Psychosomatics 28:592, 594–595, 1987

    • Search Google Scholar
    • Export Citation
  • 9.

    Hurst AF: The bent back of soldiers. Br Med J 2:621623, 1918 Hurst AF: The bent back of soldiers. Br Med J 2:621–623, 1918

  • 10.

    Karbowski K: The old and the new camptocormia. Spine 24:14941498, 1999 Karbowski K: The old and the new camptocormia. Spine 24:1494–1498, 1999

    • Search Google Scholar
    • Export Citation
  • 11.

    Krack P, & Vercueil L: Review of the functional surgical treatment of dystonia. Eur J Neurol 8:389399, 2001 Krack P, Vercueil L: Review of the functional surgical treatment of dystonia. Eur J Neurol 8:389–399, 2001

    • Search Google Scholar
    • Export Citation
  • 12.

    Krauss JK, , Pohle T, & Weber S, et al. : Bilateral stimulation of globus pallidus internus for treatment of cervical dystonia. Lancet 354:837838, 1999 Krauss JK, Pohle T, Weber S, et al: Bilateral stimulation of globus pallidus internus for treatment of cervical dystonia. Lancet 354:837–838, 1999

    • Search Google Scholar
    • Export Citation
  • 13.

    Kumar R, , Lozano AM, & Sime E, et al. : Comparative effects of unilateral and bilateral subthalamic nucleus deep brain stimulation. Neurology 53:561566, 1999 Kumar R, Lozano AM, Sime E, et al: Comparative effects of unilateral and bilateral subthalamic nucleus deep brain stimulation. Neurology 53:561–566, 1999

    • Search Google Scholar
    • Export Citation
  • 14.

    Lang AE: Surgical treatment of dystonia. Adv Neurol 78:185198, 1998 Lang AE: Surgical treatment of dystonia. Adv Neurol 78:185–198, 1998

    • Search Google Scholar
    • Export Citation
  • 15.

    Laroche M, , Delisle MB, & Aziza R, et al. : Is camptocormia a primary muscular disease? Spine 20:10111016, 1995 Laroche M, Delisle MB, Aziza R, et al: Is camptocormia a primary muscular disease? Spine 20:1011–1016, 1995

    • Search Google Scholar
    • Export Citation
  • 16.

    Lenz FA, , Suarez JI, & Metman LV, et al. : Pallidal activity during dystonia: somatosensory reorganisation and changes with severity. J Neurol Neurosurg Psychiatry 65:767770, 1998 Lenz FA, Suarez JI, Metman LV, et al: Pallidal activity during dystonia: somatosensory reorganisation and changes with severity. J Neurol Neurosurg Psychiatry 65:767–770, 1998

    • Search Google Scholar
    • Export Citation
  • 17.

    Lezak MD: Neuropsychological Assessment, ed 3. New York: Oxford University Press, 1995 Lezak MD: Neuropsychological Assessment, ed 3. New York: Oxford University Press, 1995

    • Search Google Scholar
    • Export Citation
  • 18.

    Liu X, , Hayward G, & Ford-Dunn H, et al. : Intraoperative recording of field potentials for assisting localisation of the subthalamic nucleus. Acta Neurochir 142:1204, 2000 (Abstract) Liu X, Hayward G, Ford-Dunn H, et al: Intraoperative recording of field potentials for assisting localisation of the subthalamic nucleus. Acta Neurochir 142:1204, 2000 (Abstract)

    • Search Google Scholar
    • Export Citation
  • 19.

    Marsden JF, , Ashby P, & Limousin-Dowsey P, et al. : Coherence between cerebellar thalamus, cortex and muscle in man: cerebellar thalamus interactions. Brain 123:14591470, 2000 Marsden JF, Ashby P, Limousin-Dowsey P, et al: Coherence between cerebellar thalamus, cortex and muscle in man: cerebellar thalamus interactions. Brain 123:1459–1470, 2000

    • Search Google Scholar
    • Export Citation
  • 20.

    Massa E, & Slater CB: Camptocormia and depression: a case report. Mil Med 154:352355, 1989 Massa E, Slater CB: Camptocormia and depression: a case report. Mil Med 154:352–355, 1989

    • Search Google Scholar
    • Export Citation
  • 21.

    Miller RW, & Forbes JF: Camptocormia. Mil Med 155:561565, 1990 Miller RW, Forbes JF: Camptocormia. Mil Med 155:561–565, 1990

  • 22.

    Nandi D, , Liu X, & Bain P, et al. : Electrophysiological confirmation of the zona incerta as a target for surgical treatment of disabling involuntary arm movements in multiple sclerosis: use of local field potentials. J Clin Neurosci 9:6468, 2002 Nandi D, Liu X, Bain P, et al: Electrophysiological confirmation of the zona incerta as a target for surgical treatment of disabling involuntary arm movements in multiple sclerosis: use of local field potentials. J Clin Neurosci 9:64–68, 2002

    • Search Google Scholar
    • Export Citation
  • 23.

    Nieves AV, , Miyasaki JM, & Lang AE: Acute onset dystonic camptocormia caused by lenticular lesions. Mov Disord 16:177180, 2001 Nieves AV, Miyasaki JM, Lang AE: Acute onset dystonic camptocormia caused by lenticular lesions. Mov Disord 16:177–180, 2001

    • Search Google Scholar
    • Export Citation
  • 24.

    Pahwa R, , Lyons K, & Koller WC: Surgical treatment of essential tremor. Neurology 54 (Suppl 4):S39S44, 2000 Pahwa R, Lyons K, Koller WC: Surgical treatment of essential tremor. Neurology 54 (Suppl 4):S39–S44, 2000

    • Search Google Scholar
    • Export Citation
  • 25.

    Papanastassiou V, , Rowe SJ, & Scott R, et al. : Use of the Radionics Image FusionTM and StereoplanTM programs for target localization in functional neurosurgery. J Clin Neurosci 5:2832, 1998 Papanastassiou V, Rowe SJ, Scott R, et al: Use of the Radionics Image FusionTM and StereoplanTM programs for target localization in functional neurosurgery. J Clin Neurosci 5:28–32, 1998

    • Search Google Scholar
    • Export Citation
  • 26.

    Parker ES, , Eaton EM, & Whipple SC, et al. : University of Southern California Repeatable Episodic Memory Test. J Clin Exp Neuropsychol 17:926936, 1995 Parker ES, Eaton EM, Whipple SC, et al: University of Southern California Repeatable Episodic Memory Test. J Clin Exp Neuropsychol 17:926–936, 1995

    • Search Google Scholar
    • Export Citation
  • 27.

    Parkin S, , Aziz T, & Gregory R, et al. : Bilateral internal globus pallidus stimulation for the treatment of spasmodic torticollis. Mov Disord 16:489493, 2001 Parkin S, Aziz T, Gregory R, et al: Bilateral internal globus pallidus stimulation for the treatment of spasmodic torticollis. Mov Disord 16:489–493, 2001

    • Search Google Scholar
    • Export Citation
  • 28.

    Reichel G, , Kirchhofer U, & Stenner A: Kamptokormia—eine segmentale Dystonie: Vorschlag einer neuen Definition fur eine alte Krankheit. Nervenarzt 72:281285, 2001 Reichel G, Kirchhofer U, Stenner A: Kamptokormia—eine segmentale Dystonie: Vorschlag einer neuen Definition fur eine alte Krankheit. Nervenarzt 72:281–285, 2001

    • Search Google Scholar
    • Export Citation
  • 29.

    Sandler SA: Camptocormia: a functional condition of the back in neurotic soldiers. War Med 8:3645, 1945 (Reference unverified) Sandler SA: Camptocormia: a functional condition of the back in neurotic soldiers. War Med 8:36–45, 1945 (Reference unverified)

    • Search Google Scholar
    • Export Citation
  • 30.

    Scott R, , Gregory R, & Hines N, et al. : Neuropsychological, neurological and functional outcome following pallidotomy for Parkinson's disease. A consecutive series of eight simultaneous bilateral and twelve unilateral procedures. Brain 121:659675, 1998 Scott R, Gregory R, Hines N, et al: Neuropsychological, neurological and functional outcome following pallidotomy for Parkinson's disease. A consecutive series of eight simultaneous bilateral and twelve unilateral procedures. Brain 121:659–675, 1998

    • Search Google Scholar
    • Export Citation
  • 31.

    Siegfried J, & Lippitz B: Bilateral chronic electrostimulation of ventroposterolateral pallidum: a new therapeutic approach for alleviating all parkinsonian symptoms. Neurosurgery 35:11261130, 1994 Siegfried J, Lippitz B: Bilateral chronic electrostimulation of ventroposterolateral pallidum: a new therapeutic approach for alleviating all parkinsonian symptoms. Neurosurgery 35:1126–1130, 1994

    • Search Google Scholar
    • Export Citation
  • 32.

    Thompson LL, & Heaton RK: Comparison of different versions of the Boston Naming Test. Clin Neuropsychol 3:184192, 1989 Thompson LL, Heaton RK: Comparison of different versions of the Boston Naming Test. Clin Neuropsychol 3:184–192, 1989

    • Search Google Scholar
    • Export Citation
  • 33.

    Tronnier VM, & Fogel W: Pallidal stimulation for generalized dystonia: Report of three cases. J Neurosurg 92:453456, 2000 Tronnier VM, Fogel W: Pallidal stimulation for generalized dystonia: Report of three cases. J Neurosurg 92:453–456, 2000

    • Search Google Scholar
    • Export Citation
  • 34.

    Trottenberg T, , Paul G, & Meissner W, et al. : Pallidal and thalamic neurostimulation in severe tardive dystonia. J Neurol Neurosurg Psychiatry 70:557559, 2001 Trottenberg T, Paul G, Meissner W, et al: Pallidal and thalamic neurostimulation in severe tardive dystonia. J Neurol Neurosurg Psychiatry 70:557–559, 2001

    • Search Google Scholar
    • Export Citation
  • 35.

    Van Gerpen JA: Camptocormia secondary to early amyotrophic lateral sclerosis. Mov Disord 16:358360, 2001 Van Gerpen JA: Camptocormia secondary to early amyotrophic lateral sclerosis. Mov Disord 16:358–360, 2001

    • Search Google Scholar
    • Export Citation
  • 36.

    Woodard JL, , Benedict RH, & Roberts VJ, et al. : Short-form alternatives to the Judgment of Line Orientation Test. J Clin Exp Neuropsychol 18:898904, 1996 Woodard JL, Benedict RH, Roberts VJ, et al: Short-form alternatives to the Judgment of Line Orientation Test. J Clin Exp Neuropsychol 18:898–904, 1996

    • Search Google Scholar
    • Export Citation
  • 37.

    Zwecker M, , Iancu I, & Zeilig G, et al. : Camptocormia: a case of possible paraneoplastic aetiology. Clin Rehabil 12:157160, 1998 Zwecker M, Iancu I, Zeilig G, et al: Camptocormia: a case of possible paraneoplastic aetiology. Clin Rehabil 12:157–160, 1998

    • Search Google Scholar
    • Export Citation

Metrics

All Time Past Year Past 30 Days
Abstract Views 273 87 15
Full Text Views 220 0 0
PDF Downloads 88 0 0
EPUB Downloads 0 0 0