Optimization of pain management in cervical dystonia

Michał Marciniec, Anna Szczepańska-Szerej, Marcin Kulczyński, Klaudia Sapko, Konrad Rejdak


Cervical dystonia (CD) is the third most common movement disorder characterized by sustained or intermittent muscle contractions causing abnormal movements, postures, or both. Pain in the course of CD is a frequent symptom reported by the 54.6% - 88.9% of patients, which strongly affects the disability and quality of life, and is the most common reason patients are looking for treatment. Despite the main effect of botulinum toxin (BoNT) is muscle relaxation through the inhibition of the acetylcholine release at the neuromuscular junction, the analgesic effect of BoNT is probably attributed to the acting on central nervous system. Up to 20% of patients discontinue therapy due to treatment failure or adverse effects. Most poor responses are related to suboptimal treatment and a minority to immunoresistance which currently concerns only 0-2,5% of CD cases. In case of confirmed immunoresistance to BoNT-A standard therapy, the use of BoNT-B or alternative BoNT-A is recommended. The currently available management of improving the analgesic efficacy of first-line treatment in patients without immunoresistance includes: the eradication of BoNT adverse events, the determination of individual BoNT dosage, reviewing injections technique with electromyography or ultrasound guidance, the implementation of a rehabilitation program and the applying of the invasive or non-invasive brain stimulation methods. However, due to the lack of evidences from the large, randomized, controlled, clinical trials, an issuance of unambiguous recommendations remains difficult. Further studies on a poor response to BoNT injections and analgesic effects of above methods in the treatment of the CD-related pain are needed.


Cervical Dystonia; Torticollis; Pain; Botulinum Toxins

Full Text:



Albanese A, Bhatia K, Bressman SB, Delong MR, Fahn S, Fung VS, et. al. Phenomenology and classification of dystonia: a consensus update. Mov Disord. 2013;28(7):863-73.

Castelão M, Marques RE, Duarte GS, Rodrigues FB, Ferreira J, Sampaio C, Moore AP, Costa J. Botulinum toxin type A therapy for cervical dystonia. Cochrane Database Syst Rev. 2017;12. https://doi.org/10.1002/14651858.CD003633.pub3.

Foley JA, Vinke RS, Limousin P, Cipolotti L. Relationship of Cognitive Function to Motor Symptoms and Mood Disorders in Patients With Isolated Dystonia. Cogn Behav Neurol. 2017;30(1):16-22.

Werle RW, Takeda SY, Zonta MB, Guimarães AT, Teive HA. The physical, social and emotional aspects are the most affected in the quality of life of the patients with cervical dystonia. Arq Neuropsiquiatr. 2014;72(6):405-10.

Avenali M, De Icco R, Tinazzi M, Defazio G, Tronconi L, Sandrini G, et al. Pain in focal dystonias - A focused review to address an important component of the disease. Parkinsonism Relat Disord. 2018. doi: 10.1016/j.parkreldis.2018.04.030.

Novak I, Campbell L, Boyce M, Fung VS. Botulinum toxin assessment, intervention and aftercare for cervical dystonia and other causes of hypertonia of the neck: international consensus statement. Eur J Neurol. 2010;2:94-108.

Kuyper DJ, Parra V, Aerts S, Okun MS, Kluger BM. Nonmotor manifestations of dystonia: a systematic review. Mov Disord. 2011;26(7):1206-17.

Smit M, Vállez García D, de Jong BM, Zoons E, Booij J, Dierckx RA, et al. Relationships between Serotonin Transporter Binding in the Raphe Nuclei, Basal Ganglia, and Hippocampus with Clinical Symptoms in Cervical Dystonia: A [11C]DASB Positron Emission Tomography Study. Front Neurol. 2018;9:88.

Albanese A, Sorbo FD, Comella C, Jinnah HA, Mink JW, Post B, et al. Dystonia rating scales: critique and recommendations. Mov Disord. 2013;28(7):874-83.

Jost WH, Hefter H, Stenner A, Reichel G. Rating scales for cervical dystonia: a critical evaluation of tools for outcome assessment of botulinum toxin therapy. J Neural Transm. 2013;120(3):487-96.

Cano SJ, Warner TT, Linacre JM, Bhatia KP, Thompson AJ, Fitzpatrick R, et al. Capturing the true burden of dystonia on patients: the Cervical Dystonia Impact Profile (CDIP-58). Neurology. 2004;63(9):1629-33.

Hawker GA, Mian S, Kendzerska T, French M. Measures of adult pain: Visual Analog Scale for Pain (VAS Pain), Numeric Rating Scale for Pain (NRS Pain), McGill Pain Questionnaire (MPQ), Short-Form McGill Pain Questionnaire (SF-MPQ), Chronic Pain Grade Scale (CPGS), Short Form-36 Bodily Pain Scale (SF-36 BPS), and Measure of Intermittent and Constant Osteoarthritis Pain (ICOAP). Arthritis Care Res. 2011;11:S240-52.

Comella CL, Fox SH, Bhatia KP, Perlmutter JS, Jinnah HA, Zurowski M, et al. Development of the Comprehensive Cervical Dystonia Rating Scale: Methodology. Mov Disord Clin Pract. 2015;2(2):135-41.

Guyatt GH, Oxman AD, Vist GE, Kunz R, Falck-Ytter Y, Alonso-Coello P, et al. GRADE: an emerging consensus on rating quality of evidence and strength of recommendations. BMJ. 2008;336(7650):924-6.

Albanese A, Asmus F, Bhatia KP, Elia AE, Elibol B, Filippini G, Gasser T, Krauss JK, Nardocci N, Newton A, Valls-Solé J. EFNS guidelines on diagnosis and treatment of primary dystonias. Eur J Neurol. 2011;18(1):5-18.

Simpson DM, Hallett M, Ashman EJ, Comella CL, Green MW, Gronseth GS, et al. 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. 2016;86: 1818-26.

Albanese A, Abbruzzese G, Dressler D, Duzynski W, Khatkova S, et al. Practical guidance for CD management involving treatment of botulinum toxin: a consensus statement. J Neurol. 2015;262(10):2201-13.

Duarte GS, Castelão M, Rodrigues FB, Marques RE, Ferreira J, Sampaio C, et al. Botulinum toxin type A versus botulinum toxin type B for cervical dystonia. Cochrane Database Syst Rev. 2016;10:CD004314.

Relja M, Miletić V. When movement disorders hurt: Addressing pain in hyperkinetic disorders. Parkinsonism Relat Disord. 2017;44:110-113.

Matak I, Lacković Z, Relja M. Botulinum toxin type A in motor nervous system: unexplained observations and new challenges. J Neural Transm (Vienna). 2016;123(12):1415-21.

Aoki KR. Review of a proposed mechanism for the antinociceptive action of botulinum toxin type A. Neurotoxicology. 2005;26(5):785-93.

Charles PD, Adler CH, Stacy M, Comella C, Jankovic J, Manack, et al. Cervical dystonia and pain: characteristics and treatment patterns from CD PROBE (Cervical Dystonia Patient Registry for Observation of OnabotulinumtoxinA Efficacy). J Neurol. 2014;261(7):1309-19.

Hefter H, Benecke R, Erbguth F, Jost W, Reichel G, Wissel J. An open-label cohort study of the improvement of quality of life and pain in de novo cervical dystonia patients after injections with 500 U botulinum toxin A (Dysport). BMJ Open. 2013;3(4).e001853.

Safarpour Y, Jabbari B. Botulinum Toxin Treatment of Movement Disorders. Curr Treat Options Neurol. 2018;20(2):4.

Duarte GS, Rodrigues FB, Ferreira JJ, Costa J. Adverse events with botulinum toxin treatment in cervical dystonia: How much should we blame placebo? Parkinsonism Parkinsonism Relat Disord. 2018. https://doi.org/10.1016/j.parkreldis.2018.06.017.

Marion MH, Humberstone M, Grunewald R, Wimalaratna S. British Neurotoxin Network recommendations for managing cervical dystonia in patients with a poor response to botulinum toxin. Pract Neurol. 2016;16(4):288-95.

Evidente VG, Pappert EJ. Botulinum toxin therapy for cervical dystonia: the science of dosing. Tremor Other Hyperkinet Mov. 2014;4:273.

Kaymak B, Kara M, Gürçay E, Özçakar L. Sonographic Guide for Botulinum Toxin Injections of the Neck Muscles in Cervical Dystonia. Phys Med Rehabil Clin N Am. 2018;29(1):105-23.

Schramm A, Bäumer T, Fietzek U, Heitmann S, Walter U, Jost WH. Relevance of sonography for botulinum toxin treatment of cervical dystonia: an expert statement. .

J Neural Transm (Vienna). 2015;122(10):1457-63.

Tamás G, Abrantes C, Valadas A, Radics P, Albanese A, Tijssen MAJ, et al. Quality and reporting of guidelines on the diagnosis and management of dystonia. Eur J Neurol. 2018;25(2):275-283.

De Bruijn E, Nijmeijer SW, Forbes PA, Koelman JH, van der Helm FC, et al. Improved identification of dystonic cervical muscles via abnormal muscle activity during isometric contractions. J Neurol Sci. 2015;354(1-2):10-6.

Wu C, Xue F, Chang W, Lian Y, Zheng Y, Xie N, et al. Botulinum toxin type A with or without needle electromyographic guidance in patients with cervical dystonia. Springerplus. 2016;5(1):1292.

Isabel Useros-Olmo A, Martínez-Pernía D, Huepe D. The effects of a relaxation program featuring aquatic therapy and autogenic training among people with cervical dystonia (a pilot study). Physiother Theory Pract. 2018. doi: 10.1080/09593985.2018.1488319.

De Pauw J1, Van der Velden K, Meirte J, Van Daele U, Truijen S, Cras P, et al. The effectiveness of physiotherapy for cervical dystonia: a systematic literature review. J Neurol. 2014;261(10):1857-65.

Tassorelli C, Mancini F, Balloni L, Pacchetti C, Sandrini G, Nappi G, et al. Botulinum toxin and neuromotor rehabilitation: An integrated approach to idiopathic cervical dystonia. Mov Disord. 2006;21(12):2240-3.

Queiroz MA, Chien HF, Sekeff-Sallem FA, Barbosa ER. Physical therapy program for cervical dystonia: a study of 20 cases. Funct Neurol. 2012;27(3):187-92.

Costa J, Espírito-Santo C, Borges A, Ferreira JJ, Coelho M, Sampaio C. Botulinum toxin type A versus anticholinergics for cervical dystonia. Cochrane Database Syst Rev. 2005;(1):CD004312.

Patel S, Martino D. Cervical dystonia: from pathophysiology to pharmacotherapy. Behav Neurol. 2013;26(4):275-82.

Mahajan A, Jankovic J, Marsh L, Patel A, Jinnah HA, Comella C, Barbano R, Perlmutter J, Patel N. Cervical dystonia and substance abuse. J Neurol. 2018;265(4):970-975.

Robinson S, Robertson FC, Dasenbrock HH, O'Brien CP, Berde C, Padua H. Image-guided intrathecal baclofen pump catheter implantation: a technical note and case series. J Neurosurg Spine. 2017;26(5):621-7.

Dykstra DD, Mendez A, Chappuis D, Baxter T, DesLauriers L, Stuckey M. Treatment of cervical dystonia and focal hand dystonia by high cervical continuously infused intrathecal baclofen: a report of 2 cases. Arch Phys Med Rehabil. 2005;86(4):830-3.

Contarino MF, Van Den Dool J, Balash Y, Bhatia K, Giladi N, Koelman JH, et al. Clinical Practice: Evidence-Based Recommendations for the Treatment of Cervical Dystonia with Botulinum Toxin. Front Neurol. 2017;8:35.

Albanese A. Deep brain stimulation for cervical dystonia. Lancet Neurol. 2014;13(9):856-7.

Skogseid IM, Ramm-Pettersen J, Volkmann J, Kerty E, Dietrichs E, Røste GK. Good long-term efficacy of pallidal stimulation in cervical dystonia: a prospective, observer-blinded study. Eur J Neurol. 2012;19(4):610-5.

Kim JP, Chang WS, Park YS, Chang JW. Effects of relative low-frequency bilateral globus pallidus internus stimulation for treatment of cervical dystonia. Stereotact Funct Neurosurg. 2012;90(1):30-6.

Angelakis E1, Liouta E, Andreadis N, Leonardos A, Ktonas P, Stavrinou LC, et al. Transcranial alternating current stimulation reduces symptoms in intractable idiopathic cervical dystonia: a case study. Neurosci Lett. 2013;533:39-43.

Bradnam LV, Frasca J, Kimberley TJ. Direct current stimulation of primary motor cortex and cerebellum and botulinum toxin a injections in a person with cervical dystonia. Brain Stimul. 2014;7(6):909-11.

DOI: http://dx.doi.org/10.5281/zenodo.1307288


  • There are currently no refbacks.

Copyright (c) 2018 © The Author(s)

Creative Commons License
This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.

Journal of Education, Health and Sport formerly Journal of Health Sciences

Declaration on the original version.

Editors indicates that the main version of the magazine is to issue a "electronic".

The journal has had 5 points in Ministry of Science and Higher Education parametric evaluation. § 8. 2) and § 12. 1. 2) 22.02.2019.

1223 Journal of Education, Health and Sport eISSN 2391-8306 7

ISSN 2391-8306 formerly ISSN: 1429-9623 / 2300-665X

Archives 2011 - 2014

PBN 2011 - 2014


POL-index 2011 - 2014


BASE 2011 - 2014




Indexed in Bases, Bazy indeksacyjne: ERIH Plus, Worldcat, PBN/POL-Index, ICI Journals Master List, Directory of Open Access Journals (DOAJ), ZBD, Ulrich's periodicals, Google Scholar, Polska Bibliografia Lekarska

US NLM = 101679844

101679844 - NLM Catalog Result - NCBI


Find a library that holds this journal: http://worldcat.org/issn/23918306

Journal Language(s): English 

PBN Poland






Redaction, Publisher and Editorial Office

Instytut Kultury Fizycznej Uniwersytet Kazimierza Wielkiego w Bydgoszczy, Institute of Physical Education Kazimierz Wielki University in Bydgoszcz, Poland 85-091 Bydgoszcz ul. Sportowa 2  www.ukw.edu.pl Copyright by Instytut Kultury Fizycznej UKW w Bydgoszczy http://ojs.ukw.edu.pl/index.php/johs  Open Access ISSN 2391-8306 formerly ISSN: 1429-9623 / 2300-665X

The journal has been approved for inclusion in ERIH PLUS.

The ERIH PLUS listing of the journal is available at https://dbh.nsd.uib.no/publiseringskanaler/erihplus/periodical/info?id=485984

SIC Science citation index (calculated on the basis of TCI and Page Rank) 0

Russian Impact factor 0.16

Indexed in Index Copernicus Journals Master List.


ICV 2018 = 95.95 ICV 2017 = 91.30 ICV 2016 = 84.69 ICV 2015 = 93.34 ICV 2014 = 89.51 Standardized Value: 8.27 ICV 2013: 7.32 ICV 2012: 6.41 ICV 20115.48

The InfoBase Index IBI Factor for the year 2015 is 3.56 in InfoBase Index.com.

Website: www.infobaseindex.com

Universal Impact Factor 1.78 for year 2012. (http://www.uifactor.org/AppliedJournals.aspx)

Indexed in Polish Scholarly Bibliography (PBN) (PBN Polska Bibliografia Naukowa) (https://pbn.nauka.gov.pl/journals/36616)

is a portal of the Polish Ministry of Science and Higher Education, collecting information on publications of Polish scientists and on Polish and foreign scholarly journals. Polish Scholarly Bibliograhpy is a part of POL-on - System of Information on Higher Education. It is operated by the Interdisciplinary Centre for Mathematical and Computational Modelling, University of Warsaw.

Indexed in Russian Sciences Index Российский индекс научного цитирования (РИНЦ) http://elibrary.ru/contents.asp?titleid=37467

Indexed in Arianta Polish scientific and professional electronic journals Aneta Drabek i Arkadiusz Pulikowski