Sensorimotor Perceptive Rehabilitation Integrated (SPRInt) program: exercises with augmented movement feedback associated to botulinum neurotoxin in idiopathic cervical dystonia—an observational study

  • Anna Castagna
  • Antonio CaronniEmail author
  • Alessandro Crippa
  • Luciana Sciumè
  • Giulia Giacobbi
  • Chiara Corrini
  • Angelo Montesano
  • Marina Ramella
Original Article



Idiopathic cervical dystonia (ICD) is a focal dystonia affecting neck muscles. Botulinum neurotoxin (BoNT) is the first-line treatment of ICD and different physical therapies (including exercise) are often proposed as adjunct treatments. However, the actual effectiveness of exercise in ICD is unclear. The aim of the current work is to assess the potential effectiveness of the Sensorimotor Perceptive Rehabilitation Integrated (SPRInt) exercise program as adjunct therapy for ICD.


Fifteen ICD patients received BoNT injections in the neck muscles and, 12 weeks later, received BoNT a second time and SPRInt started. SPRInt consists in 18 exercise sessions in which augmented feedback of movement (including visual and acoustic feedback) is extensively used. Dystonia burden was measured by the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS). Patients were evaluated immediately before, 6 and 12 weeks after each BoNT injection.


Six weeks after the first BoNT injection (i.e., at BoNT peak effect), TWSTRS total score was better than baseline and remained improved at 12 weeks. TWSTRS disability domain slightly improved 6 weeks after the first BoNT injection, but after 6 more weeks returned to its baseline level. Disability improved more at SPRInt end (i.e., 6 weeks after the second BoNT injection), being even lower than after toxin alone. With a single-subject analysis, 4/10 patients who did not improve disability after BoNT improved after SPRInt plus BoNT.


SPRInt plus BoNT can be more effective than BoNT alone in improving cervical dystonia patients’ difficulties in the activities of daily living.

Trial registration, identifier NCT03247868 (


Cervical dystonia Botulinum toxins Type A Exercise Physical therapy modalities Neurofeedback Rehabilitation 



We are particularly grateful to the patient who agreed to appear in the video attached as supplementary materials.

Funding support

This research was supported by the financial contribution of Italian Ministry of Health-Ricerca Corrente.

Compliance with ethical standards

Ethical aspects of the study were reviewed and approved by the local ethic committee (Comitato Etico “IRCCS Fondazione Don Carlo Gnocchi”, Comitato Etico Centrale IRCCS Regione Lombardia) and the study has been recorded at (NCT03247868). Each participant gave his/her written consent to take part in the study. The patient who agreed to appear in the video attached to the current work as Supplementary Materials 2 gave her written consent to publication.

Competing interests

The other authors declare that they have no conflicts of interest. Alessandro Crippa created the feedback system used in the study (Leonardo, produced by Chinesport).

Supplementary material

10072_2019_4061_MOESM1_ESM.docx (180 kb)
Supplementary Materials 1 (DOCX 180 kb)


  1. 1.
    Albanese A, Bhatia K, Bressman SB, DeLong MR, Fahn S, Fung VSC, Hallett M, Jankovic J, Jinnah HA, Klein C, Lang AE, Mink JW, Teller JK (2013) Phenomenology and classification of dystonia: a consensus update. Mov Disord 28:863–873CrossRefPubMedPubMedCentralGoogle Scholar
  2. 2.
    Zetterberg L, Lindmark B, Söderlund A, Åsenlöf P (2012) Self-perceived non-motor aspects of cervical dystonia and their association with disability. J Rehabil Med 44:950–954CrossRefPubMedGoogle Scholar
  3. 3.
    Ben-Shlomo Y, Camfield L, Warner T (2002) What are the determinants of quality of life in people with cervical dystonia? J Neurol Neurosurg Psychiatry 72:608–614CrossRefPubMedPubMedCentralGoogle Scholar
  4. 4.
    Simpson DM, Hallett M, Ashman EJ et al (2016) Practice guideline update summary: botulinum neurotoxin for the treatment of blepharospasm, cervical dystonia, adult spasticity, and headache report of the Guideline Development Subcommittee of the American Academy of Neurology. Neurology 86:1818–1826CrossRefPubMedPubMedCentralGoogle Scholar
  5. 5.
    Castelao M, Marques RE, Duarte GS, et al (2017) Botulinum toxin type A therapy for cervical dystonia. Cochrane LibrGoogle Scholar
  6. 6.
    Comella C, Bhatia K (2015) An international survey of patients with cervical dystonia. J Neurol 262:837–848CrossRefPubMedPubMedCentralGoogle Scholar
  7. 7.
    De Pauw J, der Velden K, Meirte J et al (2014) The effectiveness of physiotherapy for cervical dystonia: a systematic literature review. J Neurol 261:1857–1865CrossRefPubMedGoogle Scholar
  8. 8.
    Smania N, Corato E, Tinazzi M et al (2003) The effect of two different rehabilitation treatments in cervical dystonia: preliminary results in four patients. Funct Neurol 18:219–226PubMedGoogle Scholar
  9. 9.
    Boyce MJ, Canning CG, Mahant N, Morris J, Latimer J, Fung VSC (2013) Active exercise for individuals with cervical dystonia: a pilot randomized controlled trial. Clin Rehabil 27:226–235CrossRefPubMedGoogle Scholar
  10. 10.
    Pelosin E, Avanzino L, Marchese R, Stramesi P, Bilanci M, Trompetto C, Abbruzzese G (2013) Kinesiotaping reduces pain and modulates sensory function in patients with focal dystonia: a randomized crossover pilot study. Neurorehabil Neural Repair 27:722–731CrossRefPubMedGoogle Scholar
  11. 11.
    Delnooz CCS, Horstink MWIM, Tijssen MA, van de Warrenburg BPC (2009) Paramedical treatment in primary dystonia: a systematic review. Mov Disord Off J Mov Disord Soc 24:2187–2198CrossRefGoogle Scholar
  12. 12.
    Hill N, Frappier-Davignon L, Morrison B (1979) The periodic health examination. Can Med Assoc J 121:1193–1254Google Scholar
  13. 13.
    Tassorelli C, Mancini F, Balloni L, Pacchetti C, Sandrini G, Nappi G, Martignoni E (2006) Botulinum toxin and neuromotor rehabilitation: an integrated approach to idiopathic cervical dystonia. Mov Disord Off J Mov Disord Soc 21:2240–2243CrossRefGoogle Scholar
  14. 14.
    Kempen JH (2011) Appropriate use and reporting of uncontrolled case series in the medical literature. Am J Ophthalmol 151:7–10CrossRefPubMedPubMedCentralGoogle Scholar
  15. 15.
    Korein J, Brudny J, Grynbaum B et al (1976) Sensory feedback therapy of spasmodic torticollis and dystonia: results in treatment of 55 patients. Adv Neurol 14:375PubMedGoogle Scholar
  16. 16.
    Marsden CD (1976) The problem of adult-onset idiopathic torsion dystonia and other isolated dyskinesias in adult life (including blepharospasm, oromandibular dystonia, dystonic writer’s cramp, and torticollis, or axial dystonia). Adv Neurol 14:259–276PubMedGoogle Scholar
  17. 17.
    Shaikh AG, Zee DS, Crawford JD, Jinnah HA (2016) Cervical dystonia: a neural integrator disorder. Brain 139:2590–2599CrossRefPubMedPubMedCentralGoogle Scholar
  18. 18.
    De Pauw J, Mercelis R, Hallemans A et al (2017) Cervical sensorimotor control in idiopathic cervical dystonia: a cross-sectional study. Brain Behav 7:e00735CrossRefPubMedPubMedCentralGoogle Scholar
  19. 19.
    Young SJ, van Doornik J, Sanger TD (2011) Visual feedback reduces co-contraction in children with dystonia. J Child Neurol 26:37–43CrossRefPubMedGoogle Scholar
  20. 20.
    Anna Castagna AA (2018) Management of cervical dystonia with botulinum neurotoxins and EMG/ultrasound guidance. Neurol Clin Pract 029785aGoogle Scholar
  21. 21.
    Wissel J (2018) Towards flexible and tailored botulinum neurotoxin dosing regimens for focal dystonia and spasticity--insights from recent studies. Toxicon 147:100–106CrossRefPubMedGoogle Scholar
  22. 22.
    Scaglione F (2016) Conversion ratio between Botox®, Dysport®, and Xeomin® in clinical practice. Toxins (Basel) 8:65CrossRefGoogle Scholar
  23. 23.
    Consky ES, Basinski A, Belle L et al (1990) The Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS): assessment of validity and inter-rater reliability. Neurology 40:445Google Scholar
  24. 24.
    Lalli S, Piacentini S, Franzini A, Panzacchi A, Cerami C, Messina G, Ferré F, Perani D, Albanese A (2012) Epidural premotor cortical stimulation in primary focal dystonia: clinical and 18F-fluoro deoxyglucose positron emission tomography open study. Mov Disord 27:533–538CrossRefPubMedGoogle Scholar
  25. 25.
    Jen M-H, Kurth H, Iheanacho I, Dinet J, Gabriel S, Wasiak R, Jost WH (2014) Assessing the burden of illness from cervical dystonia using the Toronto Western Spasmodic Torticollis Rating Scale scores and health utility: a meta-analysis of baseline patient-level clinical trial data. J Med Econ 17:803–809CrossRefPubMedGoogle Scholar
  26. 26.
    Jankovic J (1994) Therapy with botulinum toxin. Marcel DekkerGoogle Scholar
  27. 27.
    Holm S (1979) A simple sequentially rejective multiple test procedure. Scand J Stat 65–70Google Scholar
  28. 28.
    Caronni A, Sciumè L (2017) Is my patient actually getting better? Application of the McNemar test for demonstrating the change at a single subject level. Disabil Rehabil 39:1341–1347CrossRefPubMedGoogle Scholar
  29. 29.
    Andrich D (1978) A rating formulation for ordered response categories. Psychometrika 43:561–573CrossRefGoogle Scholar
  30. 30.
    Masters GN (1982) A rasch model for partial credit scoring. Psychometrika 47:149–174. CrossRefGoogle Scholar
  31. 31.
    R Core Team (2017) R: a language and environment for statistical computingGoogle Scholar
  32. 32.
    Simpson DM, Blitzer A, Brashear A et al (2008) Assessment: botulinum neurotoxin for the treatment of movement disorders (an evidence-based review) Report of the Therapeutics and Technology Assessment Subcommittee of the American Academy of Neurology. Neurology 70:1699–1706CrossRefPubMedPubMedCentralGoogle Scholar
  33. 33.
    Esquenazi A, Novak I, Sheean G, Singer BJ, Ward AB (2010) International consensus statement for the use of botulinum toxin treatment in adults and children with neurological impairments--introduction. Eur J Neurol 17:1–8CrossRefPubMedGoogle Scholar
  34. 34.
    Marques RE, Duarte GS, Rodrigues FB, et al (2016) Botulinum toxin type B for cervical dystonia. Cochrane LibrGoogle Scholar
  35. 35.
    Yiannakopoulou E (2015) Serious and long-term adverse events associated with the therapeutic and cosmetic use of botulinum toxin. Pharmacology 95:65–69CrossRefPubMedGoogle Scholar
  36. 36.
    Jankovic J, Truong D, Patel AT, Brashear A, Evatt M, Rubio RG, Oh CK, Snyder D, Shears G, Comella C (2018) Injectable DaxibotulinumtoxinA in cervical dystonia: a phase 2 dose-escalation multicenter study. Mov Disord Clin Pract 5:273–282CrossRefPubMedPubMedCentralGoogle Scholar
  37. 37.
    Jankovic J, Truong D, Patel A, et al (2018) Outcomes of week-24 completers and subjects who had follow-up beyond week 24 after a single treatment of DaxibotulinumtoxinA for injection (RT002): results of a phase 2, open-label (level II), dose escalating study in isolated cervical dystonia (P5. 037)Google Scholar
  38. 38.
    Zetterberg L, Aquilonius S-M, Lindmark B (2009) Impact of dystonia on quality of life and health in a Swedish population. Acta Neurol Scand 119:376–382CrossRefPubMedGoogle Scholar
  39. 39.
    Defazio G, Esposito M, Abbruzzese G, Scaglione CL, Fabbrini G, Ferrazzano G, Peluso S, Pellicciari R, Gigante AF, Cossu G, Arca R, Avanzino L, Bono F, Mazza MR, Bertolasi L, Bacchin R, Eleopra R, Lettieri C, Morgante F, Altavista MC, Polidori L, Liguori R, Misceo S, Squintani G, Tinazzi M, Ceravolo R, Unti E, Magistrelli L, Coletti Moja M, Modugno N, Petracca M, Tambasco N, Cotelli MS, Aguggia M, Pisani A, Romano M, Zibetti M, Bentivoglio AR, Albanese A, Girlanda P, Berardelli A (2017) The Italian Dystonia Registry: rationale, design and preliminary findings. Neurol Sci 38:819–825CrossRefPubMedGoogle Scholar
  40. 40.
    Poliziani M, Koch M, Liu X (2016) Striving for more good days: patient perspectives on botulinum toxin for the treatment of cervical dystonia. Patient Prefer Adherence 10:1601CrossRefPubMedPubMedCentralGoogle Scholar

Copyright information

© Fondazione Società Italiana di Neurologia 2019

Authors and Affiliations

  1. 1.U. O. di Recupero e Rieducazione FunzionaleI.R.C.C.S. Fondazione Don Carlo Gnocchi OnlusMilanItaly
  2. 2.U. O. di Medicina riabilitativa e neuroriabilitazioneOspedale NiguardaMilanItaly

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