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Submitted: 20 February 2013 Modified: 23 June 2017
HERDIN Record #: PCHRD13022016393853

Botulinum toxin-A targeted for focal/ multifocal dystonias- An open- label application in X-linked Dystonia-Parkinsonism.

Raymond L. Rosales,
Raymond L. Rosales,
Arlene R. Ng,
Mary Mildred Delgado-Delos Santos,
Arlene R. Ng,
Criscely L. Go,
Michelle Joya-Tanglao,
Rosalia Teleg,
Marita Dantes

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Targeted for relief of spasms, posturing, pain, impaired function and disfigurement, botulinum toxin type-A (BoNT-A) was injected in dystonias of X-linked dystonia-parkinsonism (XDP). From 1992-2012, focal/ multifocal dystonia combinations were injected in XDP at the following regions: Peri-ocular (21 cases), oromandibular (50 cases), ligual (35 cases), laryngeal (5 cases), cervical (56 cases), truncalaxil (24 cases) upper limbs (13 cases) and lower limbs (18 cases). Pain was frequently reported in 40/50 cases with oromandibular dystonia, 28/56 cases with cervical dystonia, 18/24 cases with truncal-axil dystonia and 16/31 cases with limb dystonia. Outcomes were assessment through the global dystonia rating scale (DRS) at week 4, VAS pain reduction at week 4, duration of BoNT-A effects and safety. Cranial, laryngeal and cervical dystonia showed substantial improvement (DRS median score of 3-4), whereas truncal-axil and limb dystonias showed moderate improvement (DRS median score of 2), following BoNT-A. Pain reduction ranged from 30-100% (VAS), for those dystonias that reported co-morbid pain. BoNT-A effects had a duration ranging from 8-20 weeks. Procedures were generally well tolerated, and the adverse events were most significant in laryngeal injections (voice breathiness, but was eventually followed by a strong voice). The other events were mouth dryness, dysphagia and weekness in oromandibular, cervical and limb dystonias, respectively. Therefore, BoNT-A is a safe and valuable therapeutic option for the dystonias of XDP, especially the disabling and painful dystonias. BoNT-A injection working protocols could be adopted in dystonia that adheres to cost minimization (e.g. lower dose end per selected muscles), yet achieving a substantial benefit, and a reduced adverse event profile. Futhermore, this present study allowed us to recommend a "high potency, low dillution" of BoNT-A in oromandibular, linual, laryngeal, cervical and distal limb dystonias. In dystonias of the abdominal, paraspinal and proximal limb muscles, the "low potency, high dilution" BoNT-A injection protocol could be adopted.

Publication Type
Journal
Publication Sub Type
Journal Article, Original
Title
The Philippine Journal of Neurology
Frequency
Semi-Annual
Publication Date
August 2012
Volume
16
Issue
1
Page(s)
37-45
LocationLocation CodeAvailable FormatAvailability
Philippine Council for Health Research and Development Library Box no.54 Fulltext Print Format (Request Document)
1. Rosales, R L, Bigalke, H , Dressler, D . "Pharmacology of botulinum toxin: diffrences between type A preparations." Eur J Neurol 13, 2-10, 2006
2. Pickett, A , Rosales, R L. "New Trends in the Science of Botulinum Toxin-A as applied in Dystonia." Int J Neurosci 121, 22-34, 2011
3. Costa, J , Espirito-Santo, C C, Borges, A A, Ferreira, J , Coelho, M M, Moore, P , et, al . "Botulinum toxin type A therapy for blepharospasm." Cochrane DB Syst Rev DOl: 10.1002/14651858.CD004900.pub2. , 2005
4. Costa, J , Espirito-Santo, C C, Borges, A A, Ferreira, J , Coelho, M M, Moore, P , et, al . "Botulinum toxin type A therapy for cervical dystonia." Cochrane DB Syst Rev DOl: 10.1002/14651858.CD003633.pub2. , 2005
5. Simpson, D M, Blitzer, A , Brashear, A , Comella, C , Dubinsky, R , Hallett, M , et, al . "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(19): 1699-1706, 2008
6. Albanese, A , Asmus, F , Bhatia, K P, Elia, A E, Elibol, B , Filippini, G , et, al . "EFNS guidelines on diagnosis ane treatment of primary dystonias." Eur J Neurol 18(1): 5-18, 2010
7. Novak, I , Campbell, L , Boycec, M , Fung, VS C. "Botulinum toxin assessment, intervention and aftercare for cervical dystonia and other causes of hypertonic of the neck: international consensus statement." Eur J Neurol 17, S94-108, 2010
8. Costa, J , Espirito-Santo, C C, Borges, A A, Moore, P , Ferreira, J , Coelho, M M, et, al . "Botulinum toxin type A versus anticholinergics for cervical dystonia." Cochrane DB Syst Rev DOl: 10.1002/14651858. CD004312.pub2. , 2005
9. Rosales, R L, Ng, A R, Delos Santos, M M, Fernandez, H H. "The broadening application of chemodenervation in x-linked dystonia-parkinsonism (part II): an opennlabel experience with botulinum toxin-A (Dysport) injections for oromandibular, lingual, and truncal-axial dysronias." Int J Neurosci 21, 44-56, 2011
10. Rosales, R L, Delos Santos, M M, Ng, A R, Teleg, R , Dantes, M , Lee, L V, et, al . "The broadening application of chemodenervation in x-linked dystonia-parkinsonism (part I) muscle afferent block versus botulinum toxin-A in cenrical and limb dystonias." Int J Neurosci 121, 35-43, 2011
11. Rosales, R L. "X-Linked Dystonia Parkinsonism: Clinical Phenotype, Genetics and Therapeutics." J Movement Disord 3, 32-8, 2010
12. Rosales, R L. "Dystonia, Spasticity and Botulinum Toxin Therapy: Rationale, Evidences and Clinical Context. In: Rosales RL, editor. Dystonia - The Many Facers" Rijeka Croatia: InTech , 83-93, 2012
13. Lee, L V, Rivera, C , Teleg, R , Dantes, M , Pasco, P M, Jamora, R D, et, al . "The Unique Phenomenology of Sex-Linked Dystonia Parkinsonism (XDP, DYT3, "Lubag")." Int J Neurosci 121, 3-11, 2011
14. Rosales, R L, Arimura, K , Takenaga, S , Osame, M . "Extrafusal and intrafusal muscle effects in experimental botulinum toxin-A injection." Muscle Nerve 19, 488-96, 1996
15. Rosales, R L, Dressler, D . "On muscle spindles, dystonia and botulinum toxin." Eur J Nuerol 17, 71-80, 2010
16. Curra, A , Trompetto, C , Abbruzzesse, G , Berardelli, A . "Central effects of botulinum toxin type A: Evidence and supposition." Movement Disord 19(S8): S60-4, 2004
17. Kim, D , Oh, B , Paik, N . "Central effect of botulinum toxin type A in humans." Int J Neuroscience 116, 667-80, 2006
18. Trompetto, C , Curra, A , Buccolieri, A , Suppa, A , Abbruzzesse, G , Berardelli, A . "Botulinum toxin changes intrafusal feedback in dystonia: A study with the tonic vibration reflex." Movement Disord 21(6): 777-82, 2006
19. Kanovsky, P , Rosales, R L. "Debunking the pathophysiological puzzle of dystonia: with special reference to botulinum toxin therapy." Parkinsonism Relat D 17, S11-4, 2011
20. Bhidayasiri, R , Cardoso, F , Truong, D D. "Botulinum toxin in blepharospasm and oromandibular dystonia: Comparing different botulinum toxin preparations." Eur J Neurol 13, 21-9, 2006
21. Go, C L, Rosales, R L, Caraos, R , Fernandez, H H. "The Current prevalence and factors associated with tardive dyskinesias among Filipino schizophrenic patients." Parkinsonism Relat D 15, 655-9, 2009
22. Barbano, R . "Anatomic principles for botulinum toxin injection. In: Stacy Mark, editor. Neurological Disease and Therapy: Handbook of Dystonia." New York: Informa Healthcare , 317-41, 2007
23. Hawthorne, M , Anwar, K . "The use of botulinum toxin in otolaryngology. In: Ward AB, Barnes Mp, editors. Clinical uses of botulinum toxins." UK: Cambridge University Press , 111-21, 2007
24. Kasravi, N , Jog, M S. "Botulinum toxin in the treatment of lingual movement disorders." Movement Disord 24, 2199-2202, 2009
25. Truong, D D, Bhidayasiri, R . "Botulinum toxin therapy of laryngeal muscle hyperactivity syndromes: comparing different botulinum toxin preparations." Eur J Neurol 13, 36-41, 2006
26. Benecke, R , Moore, P , Dressler, D , Naumann, M . "Cervical and axial dystonias. In: Moore P, Naumann M, editors. Handbook of botulinum toxin treatment 2nd ed." UK: Blackwell Science Ltd , 158-91, 2003
27. Moore, P , Naumann, M . "General and clinical aspects of treatment with botulinum toxin. In: Moore P, Naumann M. editors. Handbook of botulinum toxin treatment 2nd ed." UK: Blackwell Science Ltd , 28-75, 2003
28. Gelb, D J, Yoshimura, D M, Olney, R K, Lowenstein, D H, Aminoff, M J. "Change in the pattern of muscle activity following botulinum toxin injections for torticollis." Ann Neurol 29, 370-6, 1991
29. Valls-Sole, J , Tolosa, E S, Riber, G . "Neurophysiological observations on the effects of botulinum toxin treatment in patients with dystonic blepharospasm." J Neurol, Neurosurg Psychiatry 54, 310-3, 1991