An overview of the pharmacotherapeutics for dystonia: advances over the past decade

References

• Albanese A, Bhatia K, Bressman SB, et al. Phenomenology and classification of dystonia: a consensus update. Mov Disord. 2013 Jun 15 28(7):863–732.
   PubMed Web of Science ®Google Scholar
• Steeves TD, Day L, Dykeman J, et al. The prevalence of primary dystonia: a systematic review and meta-analysis. Mov Disord. 2012 Dec;27(14):1789–1796.
   PubMed Web of Science ®Google Scholar
• Lim VK. Health related quality of life in patients with dystonia and their caregivers in New Zealand and Australia. Mov Disord. 2007 May 15;22(7):998–1003.
   PubMed Web of Science ®Google Scholar
• Junker J, Berman BD, Hall J, et al. Quality of life in isolated dystonia: non-motor manifestations matter. J Neurol Neurosurg Psychiatry. 2021 Feb 9;92(6):622–628.
   PubMed Web of Science ®Google Scholar
• Molho ES, Stacy M, Gillard P, et al. Impact of cervical dystonia on work productivity: an analysis from a patient registry. Mov Disord Clin Pract. 2016 Mar-Apr;3(2):130–138.
   PubMedGoogle Scholar
• Ray S, Pal PK, Yadav R. Non-motor symptoms in cervical dystonia: a review. Ann Indian Acad Neurol. 2020 Jul-Aug;23(4):449–457.
   PubMed Web of Science ®Google Scholar
• Termsarasab P, Thammongkolchai T, Frucht SJ. Medical treatment of dystonia. J Clin Mov Disord. 2016;3(1):19.
   PubMedGoogle Scholar
• Bledsoe IO, Viser AC, San Luciano M. Treatment of dystonia: medications, neurotoxins, neuromodulation, and rehabilitation. Neurotherapeutics. 2020 Oct;17(4):1622–1644.
   PubMed Web of Science ®Google Scholar
• Quartarone A, Hallett M. Emerging concepts in the physiological basis of dystonia. Mov Disord. 2013 Jun 15;28(7):958–967.
   PubMed Web of Science ®Google Scholar
• Brüggemann N. Contemporary functional neuroanatomy and pathophysiology of dystonia. J Neural Transm (Vienna). 2021 Apr;128(4):499–508.
   PubMed Web of Science ®Google Scholar
• Richter F, Richter A. Genetic animal models of dystonia: common features and diversities. Prog Neurobiol. 2014 Oct;121:91–113.
   PubMed Web of Science ®Google Scholar
• Herzog R, Weissbach A, Bäumer T, et al. Complex dystonias: an update on diagnosis and care. J Neural Transm (Vienna). 2021 Apr;128(4):431–445.
   PubMed Web of Science ®Google Scholar
• Choudhury S, Baker MR, Chatterjee S, et al. Botulinum toxin: an update on pharmacology and newer products in development. Toxins (Basel) 2021Jan 14 13(1):58.
   PubMed Web of Science ®Google Scholar
• Jankovic J, Vuong KD, Ahsan J. Comparison of efficacy and immunogenicity of original versus current botulinum toxin in cervical dystonia. Neurology. 2003 Apr 8;60(7):1186–1188.
   PubMed Web of Science ®Google Scholar
• Arezzo JC. NeuroBloc/Myobloc: unique features and findings. Toxicon. 2009 Oct;54(5):690–696.
   PubMed Web of Science ®Google Scholar
• Spiegel LL, Ostrem JL, Bledsoe IO. FDA approvals and consensus guidelines for botulinum toxins in the treatment of dystonia. Toxins (Basel). 2020 May 17;12(5):332.
   Web of Science ®Google Scholar
• Dressler D, Pan L, Su J, et al. Lantox-the Chinese botulinum toxin drug-complete English bibliography and comprehensive formalised literature review. Toxins (Basel). 2021 May 22;13(6):370.
   PubMed Web of Science ®Google Scholar
• Yang GH, Jung HH. A new botulinum toxin potentially bioequivalent to onabotulinumtoxinA: are there any differences at all? Dermatol Surg. 2013 Jan;39(1 Pt 2):165–170.
   PubMed Web of Science ®Google Scholar
• Frevert J, Ahn KY, Park MY, et al. Comparison of botulinum neurotoxin type A formulations in Asia. Clin Cosmet Investig Dermatol. 2018;11:327–331.
   PubMed Web of Science ®Google Scholar
• Solish N, Carruthers J, Kaufman J, et al. Overview of daxibotulinumtoxina for injection: a novel formulation of botulinum toxin type A. Drugs. 2021 Dec;81(18):2091–2101.
   PubMed Web of Science ®Google Scholar
• Charles D, Brashear A, Hauser RA, et al. Efficacy, tolerability, and immunogenicity of onabotulinumtoxina in a randomized, double-blind, placebo-controlled trial for cervical dystonia. Clin Neuropharmacol. 2012 Sep-Oct;35(5):208–214.
   PubMed Web of Science ®Google Scholar
• Jankovic J, Adler CH, Charles D, et al. Primary results from the cervical dystonia patient registry for observation of onabotulinumtoxina efficacy (CD PROBE). J Neurol Sci. [2015 Feb 15];349(1–2):84–93.
   PubMed Web of Science ®Google Scholar
• Marsh WA, Monroe DM, Brin MF, et al. Systematic review and meta-analysis of the duration of clinical effect of onabotulinumtoxinA in cervical dystonia. BMC Neurol. 2014 Apr 27;14:91.
   PubMed Web of Science ®Google Scholar
• Pandey S, Sharma S. Botulinum toxin in Meige’s syndrome: a video-based case series. Neurol India. 2018 Jan-Feb;66(1):71–76.
   PubMed Web of Science ®Google Scholar
• Lew MF, Brashear A, Dashtipour K, et al. A 500 U/2 mL dilution of abobotulinumtoxinA vs. placebo: randomized study in cervical dystonia. Int J Neurosci. 2018 Jul;128(7):619–626.
   PubMed Web of Science ®Google Scholar
• Hauser RA, Truong D, Hubble J, et al. AbobotulinumtoxinA (Dysport) dosing in cervical dystonia: an exploratory analysis of two large open-label extension studies. J Neural Transm (Vienna). 2013 Feb;120(2):299–307.
   PubMed Web of Science ®Google Scholar
• Trosch RM, Espay AJ, Truong D, et al. Multicenter observational study of abobotulinumtoxinA neurotoxin in cervical dystonia: the ANCHOR-CD registry. J Neurol Sci. 2017 May 15;376:84–90.
   PubMed Web of Science ®Google Scholar
• Mordin M, Masaquel C, Abbott C, et al. Factors affecting the health-related quality of life of patients with cervical dystonia and impact of treatment with abobotulinumtoxinA (Dysport): results from a randomised, double-blind, placebo-controlled study. BMJ Open. [2014 Oct 16];4(10):e005150.
   PubMed Web of Science ®Google Scholar
• Colosimo C, Charles D, Misra VP, et al. Cumulative effects of long-term treatment with abobotulinumtoxinA in cervical dystonia: findings from a prospective, observational study. J Neurol Sci. 2020 Sep 15;416:117015.
   PubMed Web of Science ®Google Scholar
• Mitsikostas DD, Dekundy A, Hanschmann A, et al. Duration and onset of effect of incobotulinumtoxinA for the treatment of blepharospasm in botulinum toxin-naïve subjects. Curr Med Res Opin. 2021 Oct;37(10):1761–1768.
   PubMed Web of Science ®Google Scholar
• Mitsikostas DD, Dekundy A, Sternberg K, et al. IncobotulinumtoxinA for the treatment of blepharospasm in toxin-naïve subjects: a multi-center, double-blind, randomized, placebo-controlled trial. Adv Ther. 2020 Oct;37(10):4249–4265.
   PubMed Web of Science ®Google Scholar
• Dressler D, Paus S, Seitzinger A, et al. Long-term efficacy and safety of incobotulinumtoxinA injections in patients with cervical dystonia. J Neurol Neurosurg Psychiatry. 2013 Sep;84(9):1014–1019.
   PubMed Web of Science ®Google Scholar
• Fernandez HH, Pappert EJ, Comella CL, et al. Efficacy and safety of incobotulinumtoxina in subjects previously treated with botulinum toxin versus toxin-naïve subjects with cervical dystonia. Tremor Other Hyperkinet Mov (N Y). 2013 May 7;3:tre-03-140-2921-1.
   Google Scholar
• Chinnapongse RB, Lew MF, Ferreira JJ, et al. Immunogenicity and long-term efficacy of botulinum toxin type B in the treatment of cervical dystonia: report of 4 prospective, multicenter trials. Clin Neuropharmacol. 2012 Sep-Oct;35(5):215–223.
   PubMed Web of Science ®Google Scholar
• Kaji R, Endo A, Sugawara M, et al. Efficacy of botulinum toxin type B (rimabotulinumtoxinB) in patients with cervical dystonia previously treated with botulinum toxin type A: a post-marketing observational study in Japan. eNeurologicalSci. 2021;25:100374.
   PubMedGoogle Scholar
• Tsui JK, Eisen A, Mak E, et al. A pilot study on the use of botulinum toxin in spasmodic torticollis. Can J Neurol Sci. 1985 Nov;12(4):314–316.
   PubMed Web of Science ®Google Scholar
• Hefter H, Samadzazeh S, Rosenthal D. The impact of the initial severity on later outcome: retrospective analysis of a large cohort of botulinum toxin naïve patients with idiopathic cervical dystonia. J Neurol. 2021 Jan;268(1):206–213.
   PubMed Web of Science ®Google Scholar
• Lee A, Al-Sarea J, Altenmüller E. Nonlinear changes in botulinum toxin treatment of task-specific dystonia during long-term treatment. Toxins (Basel). 2021 May 22;13(6):371.
   Web of Science ®Google Scholar
• Jochim A, Meindl T, Mantel T, et al. Treatment of cervical dystonia with abo- and onabotulinumtoxinA: long-term safety and efficacy in daily clinical practice. J Neurol. 2019 Aug;266(8):1879–1886.
   PubMed Web of Science ®Google Scholar
• Jochim A, Meindl T, Huber C, et al. Treatment of blepharospasm and Meige’s syndrome with abo- and onabotulinumtoxinA: long-term safety and efficacy in daily clinical practice. J Neurol. 2020 Jan;267(1):267–275.
   PubMed Web of Science ®Google Scholar
• Kollewe K, Mohammadi B, Köhler S, et al. Blepharospasm: long-term treatment with either Botox®, Xeomin® or Dysport®. J Neural Transm (Vienna). 2015 Mar;122(3):427–431.
   PubMed Web of Science ®Google Scholar
• Dressler D, Tacik P, Adib Saberi F. Botulinum toxin therapy of cervical dystonia: comparing onabotulinumtoxinA (Botox(®)) and incobotulinumtoxinA (Xeomin (®)). J Neural Transm (Vienna). 2014 Jan;121(1):29–31.
   PubMed Web of Science ®Google Scholar
• Saad J, Gourdeau A. A direct comparison of onabotulinumtoxina (Botox) and IncobotulinumtoxinA (Xeomin) in the treatment of benign essential blepharospasm: a split-face technique. J Neuroophthalmol. 2014 Sep;34(3):233–236.
   PubMed Web of Science ®Google Scholar
• Chundury RV, Couch SM, Holds JB. Comparison of preferences between onabotulinumtoxinA (Botox) and incobotulinumtoxinA (Xeomin) in the treatment of benign essential blepharospasm. Ophthalmic Plast Reconstr Surg. 2013 May-Jun;29(3):205–207.
   PubMed Web of Science ®Google Scholar
• Lee S, Park S, Lew H. Long-term efficacy of botulinum neurotoxin-a treatment for essential blepharospasm. Korean J Ophthalmol. 2018 Feb;32(1):1–7.
   PubMedGoogle Scholar
• Sane S, Ali MJ, Naik MN. Comparison of safety and efficacy of botox and neuronox in the management of benign essential blepharospasm: a split-face study. Korean J Ophthalmol. 2019 Oct;33(5):430–435.
   PubMedGoogle Scholar
• Kongsaengdao S, Arayawithchanont A, Samintharapanya K, et al. Low-dose neubotulinum toxin A versus low-dose abobotulinum toxin A injection for the treatment of cervical dystonia: a multicenter, 48-week, prospective, double-blinded, randomized crossover design study. Toxins (Basel). 2021.Oct 1;13(10):694.
   Web of Science ®Google Scholar
• Jagota P, Kaewwilai L, Boonrod N, et al. Impact of neu-botulinumtoxinA on the severity and quality of life of cervical dystonia patients. Tremor Other Hyperkinet Mov (N Y). 2016;6:407.
   PubMed Web of Science ®Google Scholar
• Barbosa PM, Rodrigues GR, de Oliveira DS, et al. Comparison between dysport and prosigne in the treatment of cervical dystonia. Clin Neuropharmacol. 2015 Nov-Dec;38(6):221–226.
   PubMed Web of Science ®Google Scholar
• Rystedt A, Zetterberg L, Burman J, et al. A comparison of Botox 100 U/mL and Dysport 100 U/mL using dose conversion ratio 1: 3 and 1: 1.7 in the treatment of cervical dystonia: a double-blind, randomized, crossover trial. Clin Neuropharmacol. 2015 Sep-Oct;38(5):170–176.
   PubMed Web of Science ®Google Scholar
• Yun JY, Kim JW, Kim HT, et al. Dysport and Botox at a ratio of 2.5:1 units in cervical dystonia: a double-blind, randomized study. Mov Disord. 2015 Feb;30(2):206–213.
   PubMed Web of Science ®Google Scholar
• Bentivoglio AR, Ialongo T, Bove F, et al. Retrospective evaluation of the dose equivalence of Botox(®) and Dysport (®) in the management of blepharospasm and hemifacial spasm: a novel paradigm for a never ending story. Neurol Sci. 2012 Apr;33(2):261–267.
   PubMed Web of Science ®Google Scholar
• Rystedt A, Nyholm D, Naver H. Clinical experience of dose conversion ratios between 2 botulinum toxin products in the treatment of cervical dystonia. Clin Neuropharmacol. 2012 Nov-Dec;35(6):278–282.
   PubMed Web of Science ®Google Scholar
• Lange O, Bigalke H, Dengler R, et al. Neutralizing antibodies and secondary therapy failure after treatment with botulinum toxin type A: much ado about nothing? Clin Neuropharmacol. 2009 Jul-Aug;32(4):213–218.
   PubMed Web of Science ®Google Scholar
• Evidente VG, Truong D, Jankovic J, et al. IncobotulinumtoxinA (Xeomin®) injected for blepharospasm or cervical dystonia according to patient needs is well tolerated. J Neurol Sci. [2014 Nov 15];346(1–2):116–120.
   PubMed Web of Science ®Google Scholar
• Kent R, Robertson A, Quiñones Aguilar S, et al. Real-world dosing of onabotulinumtoxina and incobotulinumtoxina for cervical dystonia and blepharospasm: results from TRUDOSE and TRUDOSE II. Toxins (Basel). 2021 Jul 14;13(7):488.
   PubMed Web of Science ®Google Scholar
• Dressler D, Tacik P, Saberi FA. Botulinum toxin therapy of cervical dystonia: duration of therapeutic effects. J Neural Transm (Vienna). 2015 Feb;122(2):297–300.
   PubMed Web of Science ®Google Scholar
• Evidente VG, Fernandez HH, LeDoux MS, et al. A randomized, double-blind study of repeated incobotulinumtoxinA (Xeomin(®)) in cervical dystonia. J Neural Transm (Vienna). 2013 Dec;120(12):1699–1707.
   PubMed Web of Science ®Google Scholar
• Bellows S, Jankovic J. Immunogenicity associated with botulinum toxin treatment. Toxins (Basel). 2019 Aug 26;11(9):491.
   PubMed Web of Science ®Google Scholar
• Czyz CN, Burns JA, Bergstrom RE. Antibody development in patients treated long-term with onabotulinumtoxina for benign essential blepharospasm and hemifacial spasm. J Neuroophthalmol. 2021 Dec 1;41(4):e684–e687.
   PubMed Web of Science ®Google Scholar
• Hefter H, Spiess C, Rosenthal D. Very early reduction in efficacy of botulinum toxin therapy for cervical dystonia in patients with subsequent secondary treatment failure: a retrospective analysis. J Neural Transm (Vienna). 2014 May;121(5):513–519.
   PubMed Web of Science ®Google Scholar
• Hefter H, Schomaecker I, Schomaecker M, et al. The use of high initial doses of botulinum toxin therapy for cervical dystonia is a risk factor for neutralizing antibody formation-a monocentric cross-sectional pilot study. Medicina (Kaunas). 2022 Jan 7;58:(1):88.
   Web of Science ®Google Scholar
• Coleman C, Hubble J, Schwab J, et al. Immunoresistance in cervical dystonia patients after treatment with abobotulinumtoxinA. Int J Neurosci. 2012 Jul;122(7):358–362.
   PubMed Web of Science ®Google Scholar
• Ferreira JJ, Colosimo C, Bhidayasiri R, et al. Factors influencing secondary non-response to botulinum toxin type A injections in cervical dystonia. Parkinsonism Relat Disord. 2015 Feb;21(2):111–115.
   PubMed Web of Science ®Google Scholar
• Jinnah HA, Goodmann E, Rosen AR, et al. Botulinum toxin treatment failures in cervical dystonia: causes, management, and outcomes. J Neurol. 2016 Jun;263(6):1188–1194.
   PubMed Web of Science ®Google Scholar
• Ramos VF, Karp BI, Lungu C, et al. Clinical response to IncobotulinumtoxinA, after demonstrated loss of clinical response to onabotulinumtoxina and rimabotulininumtoxinb in a patient with musician’s dystonia. Mov Disord Clin Pract. 2014 Dec;1(4):383–385.
   PubMedGoogle Scholar
• Hefter H, Ürer B, Brauns R, et al. Significant long-lasting improvement after switch to incobotulinum toxin in cervical dystonia patients with secondary treatment failure. Toxins (Basel). 2022 Jan 6;14:(1):44.
   PubMed Web of Science ®Google Scholar
• Jochim A, Castrop F, Jochim B, et al. Secondary treatment failure in cervical dystonia after treatment with inco- and abobotulinumtoxinA. Parkinsonism Relat Disord. 2015 Jun;21(6):663–664.
   PubMed Web of Science ®Google Scholar
• Macdonald RL, Kelly KM. Antiepileptic drug mechanisms of action. Epilepsia. 1995;36(2):S2–12.
   PubMed Web of Science ®Google Scholar
• Li XH, Lin SC, Hu YF, et al. Efficacy of carbamazepine combined with botulinum toxin a in the treatment of blepharospasm and hemifacial spasm. Eye Sci. 2012 Dec;27(4):178–181.
   PubMedGoogle Scholar
• Naheed D, Mehta S, Goyal MK, et al. Carbamazepine responsive generalized dystonia in Fahr’s disease. Parkinsonism Relat Disord. 2017;40:78–79.
   PubMed Web of Science ®Google Scholar
• Aljabri MF, Kamal NM, Alghamdi A, et al. Favorable response to carbamazepine therapy in genetically proven myoclonus-dystonia child. Ital J Pediatr. [2021 Feb 15];47(1):33.
   PubMed Web of Science ®Google Scholar
• Sanjari Moghaddam H, Tafakhori A, Darvish H, et al. Treatment of Myoclonus-dystonia with carbamazepine. Parkinsonism Relat Disord. 2018;53:116–117.
   PubMed Web of Science ®Google Scholar
• Rissardo JP. Caprara ALF. carbamazepine, oxcarbazepine, eslicarbazepine-associated movement disorder: a literature review. Clin Neuropharmacol. 2020 May/Jun;43(3):66–80.
   PubMed Web of Science ®Google Scholar
• Lynch BA, Lambeng N, Nocka K, et al. The synaptic vesicle protein SV2A is the binding site for the antiepileptic drug levetiracetam. Proc Natl Acad Sci U S A. [2004 Jun 29];101(26):9861–9866.
   PubMed Web of Science ®Google Scholar
• Löscher W, Richter A. Piracetam and levetiracetam, two pyrrolidone derivatives, exert antidystonic activity in a hamster model of paroxysmal dystonia. Eur J Pharmacol. 2000 Mar 17;391(3):251–254.
   PubMed Web of Science ®Google Scholar
• Park JE, Srivanitchapoom P, Maurer CW, et al. Lack of efficacy of levetiracetam in oromandibular and cranial dystonia. Acta Neurol Scand. 2017 Aug;136(2):103–108.
   PubMed Web of Science ®Google Scholar
• Biton V. Clinical pharmacology and mechanism of action of zonisamide. Clin Neuropharmacol. 2007 Jul-Aug;30(4):230–240.
   PubMed Web of Science ®Google Scholar
• Salamon A, Zádori D, Horváth E, et al. Zonisamide treatment in myoclonus-dystonia. Orv Hetil. 2019 Aug;160(34):1353–1357.
   PubMed Web of Science ®Google Scholar
• Smit M, Bartels AL, van Faassen M, et al. Serotonergic perturbations in dystonia disorders-a systematic review. Neurosci Biobehav Rev. 2016;65:264–275.
   PubMed Web of Science ®Google Scholar
• Sanchez C, Reines EH, Montgomery SA. A comparative review of escitalopram, paroxetine, and sertraline: are they all alike? Int Clin Psychopharmacol. 2014 Jul;29(4):185–196.
   PubMed Web of Science ®Google Scholar
• Zoons E, Tijssen MAJ, Dreissen YEM, et al. The effect of escitalopram on central serotonergic and dopaminergic systems in patients with cervical dystonia, and its relationship with clinical treatment effects: a double-blind placebo-controlled trial. Biomolecules. 2020 Jun 8;10(6):880.
   PubMed Web of Science ®Google Scholar
• Zoons E, Booij J, Delnooz CCS, et al. Randomised controlled trial of escitalopram for cervical dystonia with dystonic jerks/tremor. J Neurol Neurosurg Psychiatry. 2018 Jun;89(6):579–585.
   PubMed Web of Science ®Google Scholar
• Odaka T, Suzuki T, Seza A, et al. Serotonin 5- HT4 receptor agonist (mosapride citrate). Nihon Rinsho. 2006 Aug;64(8):1491–1494.
   PubMedGoogle Scholar
• Piyasena IN, Jayasinghe JA. Mosapride (5HT4 agonist) in the treatment of blepharospasm. Ceylon Med J. 2014 Mar;59(1):26–27.
   PubMedGoogle Scholar
• Scheller D, Ullmer C, Berkels R, et al. The in vitro receptor profile of rotigotine: a new agent for the treatment of Parkinson’s disease. Naunyn Schmiedebergs Arch Pharmacol. 2009 Jan;379(1):73–86.
   PubMed Web of Science ®Google Scholar
• Fan X, Donsante Y, Jinnah HA, et al. Dopamine receptor agonist treatment of idiopathic dystonia: a reappraisal in humans and mice. J Pharmacol Exp Ther. 2018 Apr;365(1):20–26.
   PubMed Web of Science ®Google Scholar
• Mangiardi M, Alfano G. A clinical improvement of a idiopathic cervical dystonia in a patient treated with transdermal Rotigotine: a case-report. Acta Biomed. 2020 Sep 7;91(3):e2020078.
   PubMedGoogle Scholar
• Challman TD, Lipsky JJ. Methylphenidate: its pharmacology and uses. Mayo Clin Proc. 2000 Jul;75(7):711–721.
   PubMed Web of Science ®Google Scholar
• Price KM, Ramey NA, Richard MJ, et al. Can methylphenidate objectively provide relief in patients with uncontrolled blepharospasm? A pilot study using surface electromyography. Ophthalmic Plast Reconstr Surg. 2010 Sep-Oct;26(5):353–356.
   PubMed Web of Science ®Google Scholar
• Eftekhari K, Choe CH, Vagefi MR, et al. Oral methylphenidate for the treatment of refractory facial dystonias. Ophthalmic Plast Reconstr Surg. 2015 May-Jun;31(3):e65–6.
   PubMed Web of Science ®Google Scholar
• Maas R, Wassenberg T, Lin JP, et al. l-Dopa in dystonia: a modern perspective. Neurology. [2017 May 9];88(19):1865–1871.
   PubMed Web of Science ®Google Scholar
• Nygaard TG, Marsden CD, Duvoisin RC. Dopa-responsive dystonia. Adv Neurol. 1988;50:377–384.
   PubMedGoogle Scholar
• Lee WW, Jeon B, Kim R. Expanding the spectrum of dopa-responsive dystonia (DRD) and proposal for new definition: DRD, DRD-plus, and DRD look-alike. J Korean Med Sci. 2018 Jul 9;33(28):e184.
   Web of Science ®Google Scholar
• Kim R, Jeon B, Lee WW. A systematic review of treatment outcome in patients with dopa-responsive dystonia (DRD) and DRD-plus. Mov Disord Clin Pract. 2016 Sep-Oct;3(5):435–442.
   PubMedGoogle Scholar
• Winter B, Krämer J, Meinhardt T, et al. NR4A2 and dystonia with dopa responsiveness. Mov Disord. 2021;36(9):2203–2204.
   PubMed Web of Science ®Google Scholar
• Baschieri F, Batla A, Erro R, et al. Paroxysmal exercise-induced dystonia due to GLUT1 mutation can be responsive to levodopa: a case report. J Neurol. 2014 Mar;261(3):615–616.
   PubMed Web of Science ®Google Scholar
• Oravivattanakul S, Abboud H, Fernandez H, et al. Unusual case of levodopa-responsive camptocormia in a patient with negative dopamine transporter scan and normal DYT 5 gene. Clin Neuropharmacol. 2014 Mar-Apr;37(2):63–64.
   PubMed Web of Science ®Google Scholar
• Qiu J, Kumar KR, Watson E, et al. Dystonia responsive to dopamine: POLG mutations should be considered if sensory neuropathy is present. J Mov Disord. 2021 May;14(2):157–160.
   PubMed Web of Science ®Google Scholar
• Scherman D. Dihydrotetrabenazine binding and monoamine uptake in mouse brain regions. J Neurochem. 2006 Aug;47(2):331–339.
   Google Scholar
• Miguel R, Mendonça MD, Barbosa R, et al. Tetrabenazine in treatment of hyperkinetic movement disorders: an observational study. Ther Adv Neurol Disord. 2017 Feb;10(2):81–90.
   PubMed Web of Science ®Google Scholar
• Luciano AY, Jinnah HA, Pfeiffer RF, et al. Treatment of myoclonus-dystonia syndrome with tetrabenazine. Parkinsonism Relat Disord. 2014 Dec;20(12):1423–1426.
   PubMed Web of Science ®Google Scholar
• McCluggage HL. Changing from continuous SC to transdermal clonidine to treat dystonia in a teenage boy with end-stage leucodystrophy. BMJ Support Palliat Care. 2018;8(4):433–435.
   PubMed Web of Science ®Google Scholar
• Sayer C, Lumsden DE, Kaminska M, et al. Clonidine use in the outpatient management of severe secondary dystonia. Eur J Paediatr Neurol. 2017 Jul;21(4):621–626.
   PubMed Web of Science ®Google Scholar
• Chu EA, Byrne PJ. Pharmacologic reversal of Horner’s syndrome-related Ptosis with apraclonidine. Ear Nose Throat J. 2007 May;86(5):270, 273.
   PubMed Web of Science ®Google Scholar
• Vijayakumar D, Wijemanne S, Jankovic J. Treatment of blepharospasm with apraclonidine. J Neurol Sci. 2017 Jan 15; 372:57–59.
   PubMed Web of Science ®Google Scholar
• Turner M, Nguyen HS, Cohen-Gadol AA. Intraventricular baclofen as an alternative to intrathecal baclofen for intractable spasticity or dystonia: outcomes and technical considerations. J Neurosurg Pediatr. 2012 Oct;10(4):315–319.
   PubMed Web of Science ®Google Scholar
• Gianaris T, Holland RM, Villelli NW, et al. Prepontine placement of an intrathecal baclofen pump catheter for treatment of dystonia. Surg Neurol Int. 2021;12:477.
   PubMedGoogle Scholar
• Bonouvrié LA, Becher JG, Vles JSH, et al. The effect of intrathecal baclofen in dyskinetic cerebral palsy: the IDYS trial. Ann Neurol. 2019 Jul;86(1):79–90.
   PubMed Web of Science ®Google Scholar
• Monbaliu E, Ortibus E, De Cat J, et al. The Dyskinesia Impairment Scale: a new instrument to measure dystonia and choreoathetosis in dyskinetic cerebral palsy. Dev Med Child Neurol. 2012 Mar;54(3):278–283.
   PubMed Web of Science ®Google Scholar
• Barry MJ, VanSwearingen JM, Albright AL. Reliability and responsiveness of the Barry–Albright dystonia scale. Dev Med Child Neurol. 1999 Jun;41(6):404–411.
   PubMed Web of Science ®Google Scholar
• Ruggiero C, Meccariello G, Spennato P, et al. Early intraventricular baclofen therapy (IVB) for children with dystonic and dysautonomic storm. Childs Nerv Syst. 2019 Jan;35(1):15–18.
   PubMed Web of Science ®Google Scholar
• Méndez-Lucena C, Chacón Peña J, García-Moreno JM. Baclofeno intratecal para el tratamiento de la distonía durante el embarazo: un caso clínico. Neurología. 2016 Mar;31(2):131–132.
   PubMedGoogle Scholar
• Bahl A, Tripathi C, McMullan J, et al. Novel use of intrathecal baclofen drug delivery system for periodic focal dystonia in a teenager. Neuromodulation. 2013 May-Jun;16(3):273–275.
   PubMed Web of Science ®Google Scholar
• Nguyen K, Hoegerl C. A case of meige syndrome treated with baclofen. Cureus. 2020 Sep 21; 12(9):e10570.
   PubMed Web of Science ®Google Scholar
• Imbriani P, Sciamanna G, El Atiallah I, et al. Synaptic effects of ethanol on striatal circuitry: therapeutic implications for dystonia. FEBS J. 2022;289(19):5834–5849.
   PubMed Web of Science ®Google Scholar
• Rumbach AF, Blitzer A, Frucht SJ, et al. An open-label study of sodium oxybate in Spasmodic dysphonia. Laryngoscope. 2017 Jun;127(6):1402–1407.
   PubMed Web of Science ®Google Scholar
• Simonyan K, Frucht SJ, Blitzer A, et al. A novel therapeutic agent, sodium oxybate, improves dystonic symptoms via reduced network-wide activity. Sci Rep. 2018 Oct 31 8(1):16111.
   PubMedGoogle Scholar
• Salvà P, Costa J. Clinical pharmacokinetics and pharmacodynamics of zolpidem. Therapeutic implications. Clin Pharmacokinet. 1995 Sep;29(3):142–153.
   PubMed Web of Science ®Google Scholar
• Badillo SPJ, Jamora RDG. Zolpidem for the Treatment of Dystonia. Front Neurol. 2019;10:779.
   PubMed Web of Science ®Google Scholar
• Sciamanna G, Tassone A, Martella G, et al. Developmental profile of the aberrant dopamine D2 receptor response in striatal cholinergic interneurons in DYT1 dystonia. PLoS One. 2011;6(9):e24261.
   PubMed Web of Science ®Google Scholar
• Sciamanna G, Ponterio G, Tassone A, et al. Negative allosteric modulation of mGlu5 receptor rescues striatal D2 dopamine receptor dysfunction in rodent models of DYT1 dystonia. Neuropharmacology. 2014;85:440–450.
   PubMed Web of Science ®Google Scholar
• Martella G, Bonsi P, Imbriani P, et al. Rescue of striatal long-term depression by chronic mGlu5 receptor negative allosteric modulation in distinct dystonia models. Neuropharmacology. 2021 Jul 1;192:108608.
   PubMed Web of Science ®Google Scholar
• Therapeutics A. Addex provides update on dipraglurant blepharospasm phase 2 feasibility clinical study 2022. 2022. cited 2022 Aug 31. Available from: https://www.globenewswire.com/news-release/2022/05/27/2451728/0/en/Addex-Provides-Update-on-Dipraglurant-Blepharospasm-Phase-2-Feasibility-Clinical-Study.html
   Google Scholar
• Richter A, Löscher W, Löschmann PA. The AMPA receptor antagonist NBQX exerts antidystonic effects in an animal model of idiopathic dystonia. Eur J Pharmacol. 1993 Feb 9;231(2):287–291.
   PubMed Web of Science ®Google Scholar
• Sander SE, Richter A. Effects of intrastriatal injections of glutamate receptor antagonists on the severity of paroxysmal dystonia in the dtsz mutant. Eur J Pharmacol. 2007 Jun 1;563(1–3):102–108.
   PubMed Web of Science ®Google Scholar
• Fox SH, Swan M, Jinnah HA, et al. An open-label phase 2a study to evaluate the safety and tolerability of perampanel in cervical dystonia. Mov Disord Clin Pract. 2021;8(5):743–749.
   PubMed Web of Science ®Google Scholar
• Ravenscroft P, Brotchie J. NMDA receptors in the basal ganglia. J Anat. 2000 May;196(4):577–585.
   PubMedGoogle Scholar
• Avchalumov Y, Sander SE, Richter F, et al. Role of striatal NMDA receptor subunits in a model of paroxysmal dystonia. Exp Neurol. 2014;261:677–684.
   PubMed Web of Science ®Google Scholar
• Nencini C, De Santis V, Fusari M. Unexpected ketamine effect in a patient with cervical dystonia. J Neurol. 2021 Feb;268(2):712–713.
   PubMed Web of Science ®Google Scholar
• Blanes-Mira C, Clemente J, Jodas G, et al. A synthetic hexapeptide (Argireline) with antiwrinkle activity. Int J Cosmet Sci. 2002 Oct;24(5):303–310.
   PubMedGoogle Scholar
• Lungu C, Considine E, Zahir S, et al. Pilot study of topical acetyl hexapeptide-8 in the treatment for blepharospasm in patients receiving botulinum toxin therapy. Eur J Neurol. 2013 Mar;20(3):515–518.
   PubMed Web of Science ®Google Scholar
• Castiglioni C, Verrigni D, Okuma C, et al. Pyruvate dehydrogenase deficiency presenting as isolated paroxysmal exercise induced dystonia successfully reversed with thiamine supplementation. Case report and mini-review. Eur J Paediatr Neurol. 2015 Sep;19(5):497–503.
   PubMed Web of Science ®Google Scholar
• Costantini A, Trevi E, Pala MI, et al. Thiamine and dystonia 16. BMJ Case Rep. 2016;2016:bcr-2016-216721.
   PubMedGoogle Scholar
• Gerdeman GL, Ronesi J, Lovinger DM. Postsynaptic endocannabinoid release is critical to long-term depression in the striatum. Nat Neurosci. 2002 May;5(5):446–451.
   PubMed Web of Science ®Google Scholar
• Wu YW, Kim JI, Tawfik VL, et al. Input- and cell-type-specific endocannabinoid-dependent LTD in the striatum. Cell Rep. [2015 Jan 6];10(1):75–87.
   PubMed Web of Science ®Google Scholar
• Lim K, See YM, Lee J. A systematic review of the effectiveness of medical cannabis for psychiatric, movement and neurodegenerative disorders. Clin Psychopharmacol Neurosci. 2017 Nov 30;15(4):301–312.
   PubMed Web of Science ®Google Scholar
• Anis AF-S S, Sverdlov D, Hezi N, et al. A real-life study of Medical Cannabis effect on adults with dystonia MDS virtual congress 20212021 [cited 2022 August 29, 2022]; Abstract#: 93]. Available from: https://www.mdsabstracts.org/abstract/a-real-life-study-of-medical-cannabis-effect-on-adults-with-dystonia/
   Google Scholar
• Saft C, von Hein SM, Lücke T, et al. Cannabinoids for treatment of dystonia in Huntington’s disease. J Huntingtons Dis. 2018;7(2):167–173.
   PubMed Web of Science ®Google Scholar
• Mascia MM, Carmagnini D, Defazio G. Cannabinoids and dystonia: an issue yet to be defined. Neurol Sci. 2020 Apr;41(4):783–787.
   PubMed Web of Science ®Google Scholar
• Downs AM, Roman KM, Campbell SA, et al. The neurobiological basis for novel experimental therapeutics in dystonia. Neurobiol Dis. 2019 Oct;130:104526.
   PubMed Web of Science ®Google Scholar
• Weise D, Weise CM, Naumann M. Central effects of botulinum neurotoxin-evidence from human studies. Toxins (Basel). 2019 Jan 6;11(1):21.
   PubMedGoogle Scholar
• Hok P, Veverka T, Hluštík P, et al. The central effects of botulinum toxin in dystonia and spasticity. Toxins (Basel). 2021 Feb 17;13(2):155.
   PubMed Web of Science ®Google Scholar
• Caleo M, Restani L. Direct central nervous system effects of botulinum neurotoxin. Toxicon. 2018 Jun 1;147:68–72.
   PubMed Web of Science ®Google Scholar