Skip to main content

Thank you for visiting nature.com. You are using a browser version with limited support for CSS. To obtain the best experience, we recommend you use a more up to date browser (or turn off compatibility mode in Internet Explorer). In the meantime, to ensure continued support, we are displaying the site without styles and JavaScript.

Are there two different forms of functional dystonia? A multimodal brain structural MRI study

Abstract

This study assessed brain structural alterations in two diverse clinical forms of functional (psychogenic) dystonia (FD) – the typical fixed dystonia (FixFD) phenotype and the “mobile” dystonia (MobFD) phenotype, which has been recently described in one study. Forty-four FD patients (13 FixFD and 31 MobFD) and 43 healthy controls were recruited. All subjects underwent 3D T1-weighted and diffusion tensor (DT) magnetic resonance imaging (MRI). Cortical thickness, volumes of gray matter (GM) structures, and white matter (WM) tract integrity were assessed. Normal cortical thickness in both FD patient groups compared with age-matched healthy controls were found. When compared with FixFD, MobFD patients showed cortical thinning of the left orbitofrontal cortex, and medial and lateral parietal and cingulate regions bilaterally. Additionally, compared with controls, MobFD patients showed reduced volumes of the left nucleus accumbens, putamen, thalamus, and bilateral caudate nuclei, whereas MobFD patients compared with FixFD demonstrated atrophy of the right hippocampus and globus pallidus. Compared with both controls and MobFD cases, FixFD patients showed a severe disruption of WM architecture along the corpus callous, corticospinal tract, anterior thalamic radiations, and major long-range tracts bilaterally. This study showed different MRI patterns in two variants of FD. MobFD had alterations in GM structures crucial for sensorimotor processing, emotional, and cognitive control. On the other hand, FixFD patients were characterized by a global WM disconnection affecting main sensorimotor and emotional control circuits. These findings may have important implications in understanding the neural substrates underlying different phenotypic FD expression levels.

Access options

Rent or Buy article

Get time limited or full article access on ReadCube.

from$8.99

All prices are NET prices.

Fig. 1
Fig. 2
Fig. 3

References

  1. 1.

    Espay AJ, Lang AE. Phenotype-specific diagnosis of functional (psychogenic) movement disorders. Curr Neurol Neurosci Rep. 2015;15:32.

    PubMed  Google Scholar 

  2. 2.

    Schrag A, Trimble M, Quinn N, Bhatia K. The syndrome of fixed dystonia: an evaluation of 103 patients. Brain. 2004;127(Pt 10):2360–72.

    PubMed  Google Scholar 

  3. 3.

    Batla A, Stamelou M, Edwards MJ, Parees I, Saifee TA, Fox Z, et al. Functional movement disorders are not uncommon in the elderly. Mov Disord. 2013;28:540–3.

    PubMed  Google Scholar 

  4. 4.

    Fasano A, Valadas A, Bhatia KP, Prashanth LK, Lang AE, Munhoz RP, et al. Psychogenic facial movement disorders: clinical features and associated conditions. Mov Disord. 2012;27:1544–51.

    PubMed  PubMed Central  Google Scholar 

  5. 5.

    Ganos C, Aguirregomozcorta M, Batla A, Stamelou M, Schwingenschuh P, Munchau A, et al. Psychogenic paroxysmal movement disorders--clinical features and diagnostic clues. Park Relat Disord. 2014;20:41–46.

    Google Scholar 

  6. 6.

    Petrovic IN, Tomic A, Voncina MM, Pesic D, Kostic VS. Characteristics of two distinct clinical phenotypes of functional (psychogenic) dystonia: follow-up study. J Neurol. 2018;265:82–88.

    PubMed  Google Scholar 

  7. 7.

    Edwards MJ. Neurobiologic theories of functional neurologic disorders. Handb Clin Neurol. 2017;139:131–7.

    CAS  PubMed  Google Scholar 

  8. 8.

    Perez DL, Dworetzky BA, Dickerson BC, Leung L, Cohn R, Baslet G, et al. An integrative neurocircuit perspective on psychogenic nonepileptic seizures and functional movement disorders: neural functional unawareness. Clin EEG Neurosci. 2015;46:4–15.

    PubMed  Google Scholar 

  9. 9.

    Schrag AE, Mehta AR, Bhatia KP, Brown RJ, Frackowiak RS, Trimble MR, et al. The functional neuroimaging correlates of psychogenic versus organic dystonia. Brain. 2013;136(Pt 3):770–81.

    PubMed  PubMed Central  Google Scholar 

  10. 10.

    Aybek S, Nicholson TR, Draganski B, Daly E, Murphy DG, David AS, et al. Grey matter changes in motor conversion disorder. J Neurol Neurosurg Psychiatry. 2014;85:236–8.

    PubMed  Google Scholar 

  11. 11.

    Ristic AJ, Dakovic M, Kerr M, Kovacevic M, Parojcic A, Sokic D. Cortical thickness, surface area and folding in patients with psychogenic nonepileptic seizures. Epilepsy Res. 2015;112:84–91.

    PubMed  Google Scholar 

  12. 12.

    Vuilleumier P, Chicherio C, Assal F, Schwartz S, Slosman D, Landis T. Functional neuroanatomical correlates of hysterical sensorimotor loss. Brain. 2001;124(Pt 6):1077–90.

    CAS  PubMed  Google Scholar 

  13. 13.

    Nicholson TR, Aybek S, Kempton MJ, Daly EM, Murphy DG, David AS, et al. A structural MRI study of motor conversion disorder: evidence of reduction in thalamic volume. J Neurol Neurosurg Psychiatry. 2014;85:227–9.

    CAS  PubMed  Google Scholar 

  14. 14.

    Lee S, Allendorfer JB, Gaston TE, Griffis JC, Hernando KA, Knowlton RC, et al. White matter diffusion abnormalities in patients with psychogenic non-epileptic seizures. Brain Res. 2015;1620:169–76.

    CAS  PubMed  Google Scholar 

  15. 15.

    Gupta A, Lang AE. Psychogenic movement disorders. Curr Opin Neurol. 2009;22:430–6.

    PubMed  Google Scholar 

  16. 16.

    Comella CL, Leurgans S, Wuu J, Stebbins GT, Chmura T. Dystonia Study G. Rating scales for dystonia: a multicenter assessment. Mov Disord. 2003;18:303–12.

    PubMed  Google Scholar 

  17. 17.

    Burke RE, Fahn S, Marsden CD, Bressman SB, Moskowitz C, Friedman J. Validity and reliability of a rating scale for the primary torsion dystonias. Neurology. 1985;35:73–77.

    CAS  PubMed  Google Scholar 

  18. 18.

    Hinson VK, Cubo E, Comella CL, Goetz CG, Leurgans S. Rating scale for psychogenic movement disorders: scale development and clinimetric testing. Mov Disord. 2005;20:1592–7.

    PubMed  Google Scholar 

  19. 19.

    Folstein MF, Folstein SE, McHugh PR. “Mini-mental state”. A practical method for grading the cognitive state of patients for the clinician. J Psychiatr Res. 1975;12:189–98.

    CAS  PubMed  PubMed Central  Google Scholar 

  20. 20.

    Hamilton M. A rating scale for depression. J Neurol Neurosurg Psychiatry. 1960;23:56–62.

    CAS  PubMed  PubMed Central  Google Scholar 

  21. 21.

    Hamilton M. The assessment of anxiety states by rating. Br J Med Psychol. 1959;32:50–55.

    CAS  PubMed  Google Scholar 

  22. 22.

    Marin RS, Biedrzycki RC, Firinciogullari S. Reliability and validity of the Apathy Evaluation Scale. Psychiatry Res. 1991;38:143–62.

    CAS  PubMed  Google Scholar 

  23. 23.

    Nijenhuis ER, Spinhoven P, Van Dyck R, Van der Hart O, Vanderlinden J. The development and psychometric characteristics of the Somatoform Dissociation Questionnaire (SDQ-20). J Nerv Ment Dis. 1996;184:688–94.

    CAS  PubMed  Google Scholar 

  24. 24.

    Carlson EB, Putnam FW, Ross CA, Torem M, Coons P, Dill DL, et al. Validity of the Dissociative Experiences Scale in screening for multiple personality disorder: a multicenter study. Am J Psychiatry. 1993;150:1030–6.

    CAS  PubMed  Google Scholar 

  25. 25.

    Nichols TE, Holmes AP. Nonparametric permutation tests for functional neuroimaging: a primer with examples. Hum Brain Mapp. 2002;15:1–25.

    PubMed  Google Scholar 

  26. 26.

    Smith SM, Nichols TE. Threshold-free cluster enhancement: addressing problems of smoothing, threshold dependence and localisation in cluster inference. Neuroimage. 2009;44:83–98.

    PubMed  Google Scholar 

  27. 27.

    Voon V, Gallea C, Hattori N, Bruno M, Ekanayake V, Hallett M. The involuntary nature of conversion disorder. Neurology. 2010;74:223–8.

    CAS  PubMed  PubMed Central  Google Scholar 

  28. 28.

    de Lange FP, Toni I, Roelofs K. Altered connectivity between prefrontal and sensorimotor cortex in conversion paralysis. Neuropsychologia. 2010;48:1782–8.

    PubMed  Google Scholar 

  29. 29.

    Vuilleumier P. Brain circuits implicated in psychogenic paralysis in conversion disorders and hypnosis. Neurophysiol Clin. 2014;44:323–37.

    CAS  PubMed  Google Scholar 

  30. 30.

    Sharot T, Kanai R, Marston D, Korn CW, Rees G, Dolan RJ. Selectively altering belief formation in the human brain. Proc Natl Acad Sci USA. 2012;109:17058–62.

    CAS  PubMed  Google Scholar 

  31. 31.

    Peng D, Shi F, Li G, Fralick D, Shen T, Qiu M, et al. Surface vulnerability of cerebral cortex to major depressive disorder. PLoS ONE. 2015;10:e0120704.

    PubMed  PubMed Central  Google Scholar 

  32. 32.

    Draganski B, Moser T, Lummel N, Ganssbauer S, Bogdahn U, Haas F, et al. Decrease of thalamic gray matter following limb amputation. Neuroimage. 2006;31:951–7.

    CAS  PubMed  Google Scholar 

  33. 33.

    Waugh JL, Kuster JK, Levenstein JM, Makris N, Multhaupt-Buell TJ, Sudarsky LR, et al. Thalamic volume is reduced in cervical and laryngeal dystonias. PLoS ONE. 2016;11:e0155302.

    PubMed  PubMed Central  Google Scholar 

  34. 34.

    Akiki TJ, Averill CL, Wrocklage KM, Schweinsburg B, Scott JC, Martini B, et al. The association of PTSD symptom severity with localized hippocampus and amygdala abnormalities. Chronic Stress (Thousand Oaks) 2017;1:1–18.

  35. 35.

    Canu E, Agosta F, Filippi M. A selective review of structural connectivity abnormalities of schizophrenic patients at different stages of the disease. Schizophr Res. 2015;161:19–28.

    PubMed  Google Scholar 

  36. 36.

    Jenkins LM, Barba A, Campbell M, Lamar M, Shankman SA, Leow AD, et al. Shared white matter alterations across emotional disorders: a voxel-based meta-analysis of fractional anisotropy. Neuroimage Clin. 2016;12:1022–34.

    PubMed  PubMed Central  Google Scholar 

  37. 37.

    Catani M, Howard RJ, Pajevic S, Jones DK. Virtual in vivo interactive dissection of white matter fasciculi in the human brain. Neuroimage. 2002;17:77–94.

    PubMed  Google Scholar 

  38. 38.

    Perez DL, Barsky AJ, Daffner K, Silbersweig DA. Motor and somatosensory conversion disorder: a functional unawareness syndrome? J Neuropsychiatry Clin Neurosci. 2012;24:141–51.

    PubMed  Google Scholar 

  39. 39.

    Tomic A, Petrovic I, Pesic D, Voncina MM, Svetel M, Miskovic ND, et al. Is there a specific psychiatric background or personality profile in functional dystonia? J Psychosom Res. 2017;97:58–62.

    PubMed  Google Scholar 

  40. 40.

    Berlucchi G. Frontal callosal disconnection syndromes. Cortex. 2012;48:36–45.

    PubMed  Google Scholar 

  41. 41.

    Bonilha L, de Vries PM, Hurd MW, Rorden C, Morgan PS, Besenski N, et al. Disrupted thalamic prefrontal pathways in patients with idiopathic dystonia. Park Relat Disord. 2009;15:64–67.

    Google Scholar 

  42. 42.

    Bushnell MC, Duncan GH. Sensory and affective aspects of pain perception: is medial thalamus restricted to emotional issues? Exp Brain Res. 1989;78:415–8.

    CAS  PubMed  Google Scholar 

  43. 43.

    Devinsky O. Delusional misidentifications and duplications: right brain lesions, left brain delusions. Neurology. 2009;72:80–87.

    PubMed  Google Scholar 

Download references

Acknowledgements

The study was partially supported by the Ministry of Education and Science Republic of Serbia (grant #175090).

Author information

Affiliations

Authors

Corresponding author

Correspondence to Massimo Filippi.

Ethics declarations

Conflict of interest

AT, ES, SB, DP, MK, and AF declare that they have no conflict of interest. FA is Section Editor of NeuroImage: Clinical; has received speaker honoraria from Biogen Idec and Novartis; and receives or has received research supports from the Italian Ministry of Health, AriSLA (Fondazione Italiana di Ricerca per la SLA), and the European Research Council. IP has received speaker honoraria from Boehringer Ingelheim, GSK, El pharma, Roche and Actavis. VSK has received research grants from Ministry of Education and Science, Republic of Serbia and the Serbian Academy of Science and Arts; speaker honoraria from Novartis and Boehringer Ingelheim. MF is Editor-in-Chief of the Journal of Neurology; serves on a scientific advisory board for Teva Pharmaceutical Industries; has received compensation for consulting services and/or speaking activities from Biogen Idec, Merck-Serono, Novartis, Teva Pharmaceutical Industries; and receives research support from Biogen Idec, Teva Pharmaceutical Industries, Novartis, Italian Ministry of Health, Fondazione Italiana Sclerosi Multipla, Cure PSP, Alzheimer’s Drug Discovery Foundation (ADDF), the Jacques and Gloria Gossweiler Foundation (Switzerland), and ARiSLA (Fondazione Italiana di Ricerca per la SLA).

Electronic supplementary material

Rights and permissions

Reprints and Permissions

About this article

Verify currency and authenticity via CrossMark

Cite this article

Tomic, A., Agosta, F., Sarasso, E. et al. Are there two different forms of functional dystonia? A multimodal brain structural MRI study. Mol Psychiatry 25, 3350–3359 (2020). https://doi.org/10.1038/s41380-018-0222-2

Download citation

Further reading

Search

Quick links