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CC BY-NC-ND 4.0 · Arq Neuropsiquiatr 2018; 76(03): 183-188
DOI: 10.1590/0004-282X20180013
Article

Botulinum toxin for hereditary spastic paraplegia: effects on motor and non-motor manifestations

Toxina botulínica nas paraplegias espásticas hereditárias: efeitos nas manifestações motoras e não-motoras
Katiane R. Servelhere
1   Universidade Estadual de Campinas, Hospital de Clínicas, Departamento de Neurologia, Campinas SP, Brasil
,
Ingrid Faber
1   Universidade Estadual de Campinas, Hospital de Clínicas, Departamento de Neurologia, Campinas SP, Brasil
,
Alberto Martinez
1   Universidade Estadual de Campinas, Hospital de Clínicas, Departamento de Neurologia, Campinas SP, Brasil
,
Renato Nickel
2   Universidade Federal do Paraná, Hospital de Clínicas, Departamento de Medicina Interna, Serviço de Neurologia, Unidade de Distúrbios do Movimento, Curitiba PR, Brasil.
,
Adriana Moro
2   Universidade Federal do Paraná, Hospital de Clínicas, Departamento de Medicina Interna, Serviço de Neurologia, Unidade de Distúrbios do Movimento, Curitiba PR, Brasil.
,
Francisco M. B. Germiniani
2   Universidade Federal do Paraná, Hospital de Clínicas, Departamento de Medicina Interna, Serviço de Neurologia, Unidade de Distúrbios do Movimento, Curitiba PR, Brasil.
,
Mariana Moscovich
2   Universidade Federal do Paraná, Hospital de Clínicas, Departamento de Medicina Interna, Serviço de Neurologia, Unidade de Distúrbios do Movimento, Curitiba PR, Brasil.
,
Tatiane R. Blume
2   Universidade Federal do Paraná, Hospital de Clínicas, Departamento de Medicina Interna, Serviço de Neurologia, Unidade de Distúrbios do Movimento, Curitiba PR, Brasil.
,
Renato P. Munhoz
2   Universidade Federal do Paraná, Hospital de Clínicas, Departamento de Medicina Interna, Serviço de Neurologia, Unidade de Distúrbios do Movimento, Curitiba PR, Brasil.
,
Hélio A. G. Teive
2   Universidade Federal do Paraná, Hospital de Clínicas, Departamento de Medicina Interna, Serviço de Neurologia, Unidade de Distúrbios do Movimento, Curitiba PR, Brasil.
,
Marcondes C. França Jr
1   Universidade Estadual de Campinas, Hospital de Clínicas, Departamento de Neurologia, Campinas SP, Brasil
› Author AffiliationsSupported by: Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP) 2013/01766-7
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ABSTRACT

Motor and non-motor manifestations are common and disabling features of hereditary spastic paraplegia (HSP). Botulinum toxin type A (Btx-A) is considered effective for spasticity and may improve gait in these patients. Little is known about the effects of Btx-A on non-motor symptoms in HSP patients.

Objective To assess the efficacy of Btx-A on motor and non-motor manifestations in HSP patients.

Methods Thirty-three adult patients with a clinical and molecular diagnosis of HSP were evaluated before and after Btx-A injections.

Results Mean age was 41.7 ± 13.6 years and there were 18 women. Most patients had a pure phenotype and SPG4 was the most frequent genotype. The Btx-A injections resulted in a decrease in spasticity at the adductor muscles, and no other motor measure was significantly modified. In contrast, fatigue scores were significantly reduced after Btx-A injections.

Conclusion Btx-A injections resulted in no significant functional motor improvement for HSP, but fatigue improved after treatment.

RESUMO

Manifestações motoras e não motoras são comuns e incapacitantes nas paraparesias espásticas hereditárias (PEH). Toxina botulínica do tipo A (TB-A) é considerada eficaz no tratamento da espasticidade e pode melhorar a marcha nesses pacientes. Pouco se sabe sobre os efeitos da TB-A sobre sintomas não-motores.

Objetivo avaliar a eficácia da TB-A sobre manifestações motoras e não-motoras nas PEH.

Método trinta e três pacientes adultos com PEH foram avaliados antes e depois das aplicações de TB-A.

Resultados A média de idade foi 41,7 ± 13,6 anos e havia 18 mulheres. A maioria dos pacientes portava a forma pura e o genótipo mais comum foi SPG4. Houve diminuição da espasticidade dos músculos adutores da coxa sem melhora da marcha. A pontuação da fadiga reduziu após as injeções.

Conclusão As aplicações de TB-A não melhoraram a marcha nos pacientes mas a redução da fadiga foi significativa após o tratamento.

Keywords

botulinum toxin - spastic paraplegia - hereditary - muscle spasticity

Palavras-chave

toxinas botulínicas - paraparesia espástica hereditária - espasticidade muscular

Support

This study was supported by research grants from FAPESP and CAPES.




Publication History

Received: 31 May 2017

Accepted: 18 December 2017

Article published online:
28 August 2023

© 2023. Academia Brasileira de Neurologia. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commecial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)

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  • References

  • 1 Fink JK. Hereditary spastic paraplegia: clinico-pathologic features and emerging molecular mechanisms. Acta Neuropathol. 2013 Sep;126(3):307-28. https://doi.org/10.1007/s00401-013-1115-8
  • 2 Salinas S, Proukakis C, Crosby A, Warner TT. Hereditary spastic paraplegia: clinical features and pathogenetic mechanisms. Lancet Neurol. 2008 Dec;7(12):1127-38. https://doi.org/10.1016/S1474-4422(08)70258-8
  • 3 Lo Giudice T, Lombardi F, Santorelli FM, Kawarai T, Orlacchio A. Hereditary spastic paraplegia: clinical-genetic characteristics and evolving molecular mechanisms. Exp Neurol. 2014 Nov;261:518-39. https://doi.org/10.1016/j.expneurol.2014.06.011
  • 4 Hecht MJ, Stolze H, Auf dem Brinke M, Giess R, Treig T, Winterholler M et al. Botulinum neurotoxin type A injections reduce spasticity in mild to moderate hereditary spastic paraplegia: report of 19 cases. Mov Disord. 2008 Jan;23(2):228-33. https://doi.org/10.1002/mds.21809
  • 5 Geva-Dayan K, Domenievitz D, Zahalka R, Fattal-Valevski A. Botulinum toxin injections for pediatric patients with hereditary spastic paraparesis. J Child Neurol. 2010 Aug;25(8):969-75. https://doi.org/10.1177/0883073809356037
  • 6 Rousseaux M, Launay MJ, Kozlowski O, Daveluy W. Botulinum toxin injection in patients with hereditary spastic paraparesis. Eur J Neurol. 2007 Feb;14(2):206-12. https://doi.org/10.1111/j.1468-1331.2006.01617.x
  • 7 Riccardo M, Angela L, Angela D, Vita P, Giulio L, Pietroq F et al. Combined Treatment Fkt-Botulinum Toxin Type A (Btx-A) in Patients with Strumpell-Lorrain Disease. Curr Pharm Des. 2016;22(6):758-63. https://doi.org/10.2174/1381612822666151204001830
  • 8 Niet M, de Bot ST, van de Warrenburg BP, Weerdesteyn V, Geurts AC. Functional effects of botulinum toxin type-A treatment and subsequent stretching of spastic calf muscles: a study in patients with hereditary spastic paraplegia. J Rehabil Med. 2015 Feb;47(2):147-53. https://doi.org/10.2340/16501977-1909
  • 9 Schulte-Mattler WJ. Use of botulinum toxin A in adult neurological disorders: efficacy, tolerability and safety. CNS Drugs. 2008;22(9):725-38. https://doi.org/10.2165/00023210-200822090-00002
  • 10 Muller F, Cugy E, Ducerf C, Delleci C, Guehl D, Joseph PA et al. Safety and self-reported efficacy of botulinum toxin for adult spasticity in current clinical practice: a prospective observational study. Clin Rehabil. 2012 Feb;26(2):174-9. https://doi.org/10.1177/0269215511412799
  • 11 Servelhere KR, Faber I, Saute JA, Moscovich M, D'Abreu A, Jardim LB et al. Non-motor symptoms in patients with hereditary spastic paraplegia caused by SPG4 mutations. Eur J Neurol. 2016 Feb;23(2):408-11. https://doi.org/10.1111/ene.12839
  • 12 Compston A. Aids to the investigation of peripheral nerve injuries. Medical Research Council: Nerve Injuries Research Committee. His Majesty's Stationery Office: 1942; pp. 48 (iii) and 74 figures and 7 diagrams; with aids to the examination of the peripheral nervous system. By Michael O'Brien for the Guarantors of Brain. Saunders Elsevier: 2010; pp. [8] 64 and 94 Figures. Brain. 2010 0ct;133(10):2838-44. https://doi.org/10.1093/brain/awq270
  • 13 Bohannon RW, Smith MB. Interrater reliability of a modified Ashworth scale of muscle spasticity. Phys Ther. 1987 Feb;67(2):206-7. https://doi.org/10.1093/ptj/672.206
  • 14 Rossier P, Wade DT. Validity and reliability comparison of 4 mobility measures in patients presenting with neurologic impairment. Arch Phys Med Rehabil. 2001 Jan;82(1):9-13. https://doi.org/10.1053/apmr.2001.9396
  • 15 Schüle R, Holland-Letz T, Klimpe S, Kassubek J, Klopstock T, Mall V et al. The Spastic Paraplegia Rating Scale (SPRS): a reliable and valid measure of disease severity. Neurology. 2006 Aug;67(3):430-4. https://doi.org/10.1212/01.wnl.0000228242.53336.90
  • 16 Bertolazi AN, Fagondes SC, Hoff LS, Pedro VD, Menna Barreto SS, Johns MW. Portuguese-language version of the Epworth sleepiness scale: validation for use in Brazil. J Bras Pneumol. 2009 Sep;35(9):877-83. https://doi.org/10.1590/S1806-37132009000900009
  • 17 Pavan K, Schmidt K, Marangoni B, Mendes MF, Tilbery CP, Lianza S. [Multiple sclerosis: cross-cultural adaptation and validation of the modified fatigue impact scale]. Arq Neuropsiquiatr. 2007 Sep;65 3A:669-73. https://doi.org/10.1590/S0004-282X2007000400024
  • 18 Gorenstein C, Andrade L. Validation of a Portuguese version of the Beck Depression Inventory and the State-Trait Anxiety Inventory in Brazilian subjects. Braz J Med Biol Res. 1996 Apr;29(4):453-7.
  • 19 Ferreira KA, Teixeira MJ, Mendonza TR, Cleeland CS. Validation of brief pain inventory to Brazilian patients with pain. Support Care Cancer. 2011 Apr;19(4):505-11. https://doi.org/10.1007/s00520-010-0844-7
  • 20 Pauri F, Boffa L, Cassetta E, Pasqualetti P, Rossini PM. Botulinum toxin type-A treatment in spastic paraparesis: a neurophysiological study. J Neurol Sci. 2000 Dec;181(1-2):89-97. https://doi.org/10.1016/S0022-510X(00)00439-1
  • 21 Rousseaux M, Compère S, Launay MJ, Kozlowski O. Variability and predictability of functional efficacy of botulinum toxin injection in leg spastic muscles. J Neurol Sci. 2005 May;232(1-2):51-7. https://doi.org/10.1016/j.jns.2005.01.009
  • 22 Faber I, Branco LM, França Júnior MC. Cognitive dysfunction in hereditary spastic paraplegias and other motor neuron disorders. Dement Neuropsychol. 2016 Oct-Dec;10(4):276-9. https://doi.org/10.1590/s1980-5764-2016dn1004004
  • 23 Shehata HS, Esmail EH, Abdelalim A, El-Jaafary S, Elmazny A, Sabbah A et al. Repetitive transcranial magnetic stimulation versus botulinum toxin injection in chronic migraine prophylaxis: a pilot randomized trial. J Pain Res. 2016 Oct;9:771-7. https://doi.org/10.2147/JPR.S116671
  • 24 Attal N, Andrade DC, Adam F, Ranoux D, Teixeira MJ, Galhardoni R et al. Safety and efficacy of repeated injections of botulinum toxin A in peripheral neuropathic pain (BOTNEP): a randomised, double-blind, placebo-controlled trial. Lancet Neurol. 2016 May;15(6):555-65. https://doi.org/10.1016/S1474-4422(16)00017-X