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Movement disorders
Research paper
Psychiatric associations of adult-onset focal dystonia phenotypes
  1. Brian D Berman1,2,
  2. Johanna Junker3,4,
  3. Erika Shelton1,
  4. Stefan H Sillau1,
  5. H A Jinnah5,
  6. Joel S Perlmutter6,
  7. Alberto J Espay7,
  8. Joseph Jankovic8,
  9. Marie Vidailhet9,10,
  10. Cecilia Bonnet9,10,
  11. William Ondo11,
  12. Irene A Malaty12,
  13. Ramón Rodríguez13,
  14. William M McDonald14,
  15. Laura Marsh15,
  16. Mateusz Zurowski16,
  17. Tobias Bäumer17,
  18. Norbert Brüggemann3,4
  1. 1 Department of Neurology, University of Colorado Anschutz Medical Campus, Aurora, Colorado, USA
  2. 2 Neurology Section, VA Eastern Colorado Health Care System, Denver, Colorado, USA
  3. 3 Institute of Neurogenetics, University of Luebeck, Luebeck, Germany
  4. 4 Department of Neurology, University of Luebeck, Luebeck, Germany
  5. 5 Department of Neurology, Emory University, Atlanta, Georgia, USA
  6. 6 Departments of Neurology, Radiology, Neuroscience, Physical Therapy and Occupational Therapy, Washington University in Saint Louis School of Medicine, Saint Louis, Missouri, USA
  7. 7 Department of Neurology, University of Cincinnati, Cincinnati, Ohio, USA
  8. 8 Department of Neurology, Baylor College of Medicine, Houston, Texas, USA
  9. 9 Département de neurologie, Hôpital Pitié-Salpêtrière, Assistance Publique - Hopitaux de Paris, Paris, France
  10. 10 UPMC Univ Paris 06, Inserm U1127, CNRS UMR 7225, UM 75, ICM, F-75013, Sorbonne Universites, Paris, France
  11. 11 Department of Neurology, Houston Methodist, Houston, Texas, USA
  12. 12 Department of Neurology, Center for Movement Disorders and Neurorestoration, University of Florida, Gainesville, Florida, USA
  13. 13 Department of Internal Medicine, University of Central Florida, Orlando, Florida, USA
  14. 14 Department of Psychiatry and Behavioral Sciences, Emory University, Atlanta, Georgia, USA
  15. 15 Menninger Department of Psychiatry, Baylor College of Medicine, Houston, Texas, USA
  16. 16 Department of Psychiatry, University of Toronto, Toronto, Ontario, Canada
  17. 17 Department of Paediatric and Adult Movement Disorders and Neuropsychiatry, Institute of Neurogenetics, Lübeck, Germany
  1. Correspondence to Dr Brian D Berman, Department of Neurology, University of Colorado Anschutz Medical Campus, 12631 E. 17th Avenue, Mail Stop B-185, Aurora 80045, Colorado, USA; brian.berman{at}ucdenver.edu

Abstract

Background Depression and anxiety frequently accompany the motor manifestations of isolated adult-onset focal dystonias. Whether the body region affected when this type of dystonia first presents is associated with the severity of these neuropsychiatric symptoms is unknown.

Objectives The aim of this study was to determine whether depression, anxiety and social anxiety vary by dystonia onset site and evaluate whether pain and dystonia severity account for any differences.

Methods Patients with isolated focal dystonia evaluated within 5 years from symptom onset, enrolled in the Natural History Project of the Dystonia Coalition, were included in the analysis. Individual onset sites were grouped into five body regions: cervical, laryngeal, limb, lower cranial and upper cranial. Neuropsychiatric symptoms were rated using the Beck Depression Inventory, Hospital Anxiety and Depression Scale and Liebowitz Social Anxiety Scale. Pain was estimated using the 36-Item Short Form Survey.

Results Four hundred and seventy-eight subjects met our inclusion criteria. High levels of depression, anxiety and social anxiety occurred in all groups; however, the severity of anxiety and social anxiety symptoms varied by onset site group. The most pronounced differences were higher anxiety in cervical and laryngeal, lower anxiety in upper cranial and higher social anxiety in laryngeal. Increases in pain were associated with worse neuropsychiatric symptom scores within all groups. Higher anxiety and social anxiety in laryngeal and lower anxiety in upper cranial persisted after correcting for pain and dystonia severity.

Conclusion Anxiety and social anxiety severity vary by onset site of focal dystonia, and this variation is not explained by differences in pain and dystonia severity.

  • Isolated focal dystonia
  • anxiety
  • depression
  • pain

This is an Open Access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/

https://doi.org/10.1136/jnnp-2016-315461

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    Footnotes

    • Contributors BDB: Project conception, design, organisation and execution; recruitment and clinical assessment of subjects; statistical analysis design and oversight; manuscript drafting and revision. JJ: Project conception, design and organisation; manuscript review and critique. ES: Recruitment and clinical assessment of subjects. SHS: Statistical analysis design and execution. HAJ, JSP, AJE, JJ, MV, CB, WO, IAM, RR, TB: Recruitment and clinical assessment of subjects; manuscript review and critique. WMM, LM, MZ: Neuropsychiatric scale selection; manuscript review and critique. NB: Project conception, design and organisation; recruitment and clinical assessment of subjects; manuscript review and critique.

    • Funding This work was supported in part by grants to the Dystonia Coalition (U54NS065701/TR001456), a consortium of the Rare Diseases Clinical Research Network (RDCRN) that is supported by the Office of Rare Diseases Research (ORDR) at the National Center for Advancing Clinical and Translational Studies (NCATS) in collaboration with the National Institute for Neurological Diseases and Stroke (NINDS).

    • Competing interests None declared.

    • Patient consent All patients consent with local IRB-approved consent forms.

    • Ethics approval Internal Review Boards for all participating clinical sites.

    • Provenance and peer review Not commissioned; externally peer reviewed.