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Factors predicting protracted improvement after pallidal DBS for primary dystonia: the role of age and disease duration

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Abstract

In many patients, optimal results after pallidal deep brain stimulation (DBS) for primary dystonia may appear over several months, possibly beyond 1 year after implant. In order to elucidate the factors predicting such protracted clinical effect, we retrospectively reviewed the clinical records of 44 patients with primary dystonia and bilateral pallidal DBS implants. Patients with fixed skeletal deformities, as well as those with a history of prior ablative procedures, were excluded. The Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) scores at baseline, 1 and 3 years after DBS were used to evaluate clinical outcome. All subjects showed a significant improvement after DBS implants (mean BFMDRS improvement of 74.9% at 1 year and 82.6% at 3 years). Disease duration (DD, median 15 years, range 2–42) and age at surgery (AS, median 31 years, range 10–59) showed a significant negative correlation with DBS outcome at 1 and 3 years. A partition analysis, using DD and AS, clustered subjects into three groups: (1) younger subjects with shorter DD (n = 19, AS < 27, DD ≤ 17); (2) older subjects with shorter DD (n = 8, DD ≤ 17, AS ≥ 27); (3) older subjects with longer DD (n = 17, DD > 17, AS ≥ 27). Younger patients with short DD benefitted more and faster than older patients, who however continued to improve 10% on average 1 year after DBS implants. Our data suggest that subjects with short DD may expect to achieve a better general outcome than those with longer DD and that AS may influence the time necessary to achieve maximal clinical response.

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Abbreviations

DBS:

Deep brain stimulation

GPi:

Globus pallidus internus

BFMDRS:

Burke-Fahn-Marsden Dystonia Rating Scale

AS:

Age at surgery (in years)

DD:

Disease duration (in years)

SS:

Speech and swallowing

SY:

Subjects with disease duration ≤17 years and age at surgery <27 years

SO:

Subjects with disease duration ≤17 years and age at surgery ≥27 years

LO:

Subjects with disease duration >17 years and age at surgery ≥27 years

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Acknowledgments

This study was funded in part by a grant of the Bachmann-Strauss Dystonia & Parkinson Foundation (MT) and the Mariani Foundation for Paediatric Neurology (IUI). The Authors would like to thank Silvia Molteni and Sara Tunesi for helpful advice in the implementation of the statistical analysis.

Conflict of interest

None.

Author information

Authors and Affiliations

  1. Department of Neurology, Mount Sinai School of Medicine, New York, NY, USA

    Ioannis U. Isaias & Michele Tagliati

  2. Motion Analysis Laboratory, Department of Human Physiology, University of Milano, Via Mangiagalli 32, 20133, Milan, Italy

    Ioannis U. Isaias

  3. Department of Neurology, Christian-Albrechts-University, Kiel, Germany

    Jens Volkmann & Rene Reese

  4. Department of Neurology, Charité-University Medicine, Berlin, Germany

    Andreas Kupsch, Thomas Schönecker & Andrea A. Kühn

  5. Movement Disorder Center, Department of Neurology, Bern University Hospital and University of Bern, Bern, Switzerland

    Jean-Marc Burgunder & W. M. Michael Schüpbach

  6. Department of Neurology, University of California, San Francisco, CA, USA

    Jill L. Ostrem

  7. Department of Neurosurgery, Mount Sinai School of Medicine, New York, NY, USA

    Ron L. Alterman

  8. Department of Neurosurgery, Christian-Albrechts-University, Kiel, Germany

    Hubertus Maximilian Mehdorn

  9. Department of Neurosurgery, Medical School Hannover, Hannover, Germany

    Joachim K. Krauss

  10. Department of Neurosurgery, University of California, San Francisco, CA, USA

    Philip Starr

  11. Department of Neurology, Cedars-Sinai Medical Center, Los Angeles, CA, USA

    Michele Tagliati

Authors
  1. Ioannis U. Isaias
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  2. Jens Volkmann
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  3. Andreas Kupsch
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  4. Jean-Marc Burgunder
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  5. Jill L. Ostrem
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  6. Ron L. Alterman
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  7. Hubertus Maximilian Mehdorn
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  8. Thomas Schönecker
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  9. Joachim K. Krauss
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  10. Philip Starr
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  11. Rene Reese
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  12. Andrea A. Kühn
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  13. W. M. Michael Schüpbach
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  14. Michele Tagliati
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Correspondence to Ioannis U. Isaias.

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Isaias, I.U., Volkmann, J., Kupsch, A. et al. Factors predicting protracted improvement after pallidal DBS for primary dystonia: the role of age and disease duration. J Neurol 258, 1469–1476 (2011). https://doi.org/10.1007/s00415-011-5961-9

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  • DOI: https://doi.org/10.1007/s00415-011-5961-9

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