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Sustained benefit of painful legs moving toes syndrome with botulinum toxin type A
Journal of Clinical Neuroscience xxx (2013) xxx–xxx
Contents lists available at SciVerse ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Case Report
Sustained benefit of painful legs moving toes syndrome with botulinum toxin type A Ramon L. Rodriguez a,⇑, Hubert H. Fernandez b a b
Center for Movement Disorders and Neurorestoration, University of Florida College of Medicine, 3450 Hull Rd, 4th Floor, Gainesville, FL 32607, USA Center for Neurological Restoration, Cleveland Clinic, Cleveland, OH, USA
a r t i c l e
i n f o
Article history: Available online xxxx Keywords: Botulinum toxin Dystonia Painful Legs Moving Toes
a b s t r a c t Painful legs moving toes (PLMT) is a rare disorder characterized by an often-severe painful sensation in the legs associated with involuntary movement of the toes. The treatment can be challenging given the poor response to pharmacotherapy. We present a patient with PLMT who obtained substantial benefit in both pain and severity of involuntary movement with botulinum toxin type A injections for more than 3 years. Ó 2012 Elsevier Ltd. All rights reserved.
1. Introduction The first description of PLMT made by Spillane et al. in 1971 was about six patients suffering from pain in the legs and involuntary movement of the toes.1 Common symptoms reported include deep throbbing ache in the lower extremities and constant movement of the toes in the form of flexion, extension, abduction and adduction.1 Despite an unknown etiology, PLMT may be secondary to a variety of etiologies.2–6 The pain has been described as a ‘‘constant discomfort to intractable torment’’1,7 and unfortunately, frequently shows a poor response to pharmacologic treatment.1,8 In this manuscript, we present a patient who has had a satisfactory response to more than 3 years of therapy with botulinum toxin (BoNT).
normal. A diagnosis of PLMT was made, and given the resemblance to a dystonic phenomenon, and considering the previous failure with oral medications, we decided to treat the patient with BoNT. The patient was injected with BoNT type A (Botox, Allergan, Irvine, CA, USA) under electromyographic guidance with 25 units in the flexor hallucis brevis, 25 units in adductor hallucis and 50 units in the flexor digitorium brevis. After 2 weeks the patient reported having significantly reduced abnormal involuntary movement of the toes and the pain had decreased to only minimal levels. He was able to resume practicing sports with minor discomfort. There were no side effects reported. The benefit continued for about 11 weeks, when the discomfort reappeared. Repeated injection provided similar benefit, which has been sustained for more than 3 years following reinjection every 3 months.
2. Case report A 43-year-old man presented with constant pain and occasional spasms in the right lower extremity. The most severe pain localized in the right first toe with 2 years duration. He enjoyed running marathons and the pain was becoming a problem for him to continue running. He denied any history of trauma or surgery other than the stress that may be related to physical exertion. Treatment with multiple analgesics was ineffective. The physical exam revealed constant abduction/adduction oscillations of the right toe mainly, but was also observed in the other toes to a lesser degree. No discoloration was noted in the leg or foot and the symptoms were not present in the left lower extremity. The symptoms continued to progress and eventually he was describing difficulty and pain with ambulation. His electrophysiological studies were ⇑ Corresponding author. Tel.: +1 352 294 5401; fax: +1 352 294 5399. E-mail address: [email protected]fl.edu (R.L. Rodriguez).
3. Discussion PLMT is a therapeutic challenge. A few reports have described benefit with gabapentin,9 clonazepam10 and baclofen10 but many patients fail to obtain benefit from this and other medication. Others have benefited from more invasive procedures with varying improvement.8,11,12 Our patient reported benefit 2 weeks after injection, similar to patients with dystonia, and the duration was similar to that observed in patients with cervical dystonia.13 To our knowledge, there have been only three reports of BoNT used to treat PLMT.14 However, this report is the first to describe sustained benefit over the years with toxin treatment.14–16 Given the potential for substantial, sustained improvement in both pain and abnormal involuntary movement associated in PLMT, BoNT should be considered in those patients who fail oral therapy and before considering more invasive procedures.
0967-5868/$ - see front matter Ó 2012 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jocn.2012.06.016
Please cite this article in press as: Rodriguez RL, Fernandez HH. Sustained benefit of painful legs moving toes syndrome with botulinum toxin type A. J Clin Neurosci (2013), http://dx.doi.org/10.1016/j.jocn.2012.06.016
2
Case report / Journal of Clinical Neuroscience xxx (2013) xxx–xxx
Conflicts of interest/disclosures Dr. Ramon L. Rodriguez and Dr. Fernandez have been paid consultants for Allergan. A full list of disclosures is given in Supplementary material. Appendix A. Supplementary material Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.jocn.2012.06.016. References 1. Spillane JD, Nathan PW, Kelly RE, et al. Painful legs and moving toes. Brain 1971;94:541–56. 2. Wulff CH. Painful legs and moving toes. A report of 3 cases with neurophysiological studies. Acta Neurol Scand 1982;66:283–7. 3. Bermejo PE, Zabala JA. ‘‘Painless legs and moving toes’’ syndrome due to spinal cord compression. Eur Spine J 2008;17:S294–295. 4. Montagna P, Cirignotta F, Sacquegna T, et al. ‘‘Painful legs and moving toes’’ associated with polyneuropathy. J Neurol Neurosurg Psychiatry 1983;46:399–403.
5. Ikeda K, Deguchi K, Touge T, et al. Painful legs and moving toes syndrome associated with herpes zoster myelitis. J Neurol Sci 2004;219:147–50. 6. Papapetropoulos S, Singer C. Painless legs moving toes in a patient with Wilson’s disease. Mov Disord 2006;21:579–80. 7. Dressler D, Thompson PD, Gledhill RF, et al. The syndrome of painful legs and moving toes. Mov Disord 1994;9:13–21. 8. Okuda Y, Suzuki K, Kitajima T, et al. Lumbar epidural block for ‘painful legs and moving toes’ syndrome: a report of three cases. Pain 1998;78:145–7. 9. Villarejo A, Porta-Etessam J, Camacho A, et al. Gabapentin for painful legs and moving toes syndrome. Eur Neurol 2004;51:180–1. 10. Sandyk R. Neuroleptic-induced ‘‘painful legs and moving toes’’ syndrome: successful treatment with clonazepam and baclofen. Ital J Neurol Sci 1990;11:573–6. 11. Guieu R, Sampieri F, Pouget J, et al. Adenosine in painful legs and moving toes syndrome. Clin Neuropharmacol 1994;17:460–9. 12. Raina GB, Piedimonte F, Micheli F. Posterior spinal cord stimulation in a case of painful legs and moving toes. Stereotact Funct Neurosurg 2007;85:307–9. 13. Comella CL, Jankovic J, Shannon KM, et al. Comparison of botulinum toxin serotypes A and B for the treatment of cervical dystonia. Neurology 2005;65:1423–9. 14. Trieschmann ME, Fernandez HH. Seventeenth Annual Symposia on Etiology, Pathogenesis, and Treatment of Parkinson’s Disease and Other Movement Disorders. Mov Disord 2003;18:1082–95. 15. Eisa M, Singer C, Sengun C, et al. Treatment of painful limbs/moving extremities with botulinum toxin type A injections. Eur Neurol 2008;60:104–6. 16. Schoffer K. Painful leg moving toes treated with botulinum toxin type A: a video report. Mov Disord 2010;25:784–5.
Please cite this article in press as: Rodriguez RL, Fernandez HH. Sustained benefit of painful legs moving toes syndrome with botulinum toxin type A. J Clin Neurosci (2013), http://dx.doi.org/10.1016/j.jocn.2012.06.016
Contents lists available at SciVerse ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Case Report
Sustained benefit of painful legs moving toes syndrome with botulinum toxin type A Ramon L. Rodriguez a,⇑, Hubert H. Fernandez b a b
Center for Movement Disorders and Neurorestoration, University of Florida College of Medicine, 3450 Hull Rd, 4th Floor, Gainesville, FL 32607, USA Center for Neurological Restoration, Cleveland Clinic, Cleveland, OH, USA
a r t i c l e
i n f o
Article history: Available online xxxx Keywords: Botulinum toxin Dystonia Painful Legs Moving Toes
a b s t r a c t Painful legs moving toes (PLMT) is a rare disorder characterized by an often-severe painful sensation in the legs associated with involuntary movement of the toes. The treatment can be challenging given the poor response to pharmacotherapy. We present a patient with PLMT who obtained substantial benefit in both pain and severity of involuntary movement with botulinum toxin type A injections for more than 3 years. Ó 2012 Elsevier Ltd. All rights reserved.
1. Introduction The first description of PLMT made by Spillane et al. in 1971 was about six patients suffering from pain in the legs and involuntary movement of the toes.1 Common symptoms reported include deep throbbing ache in the lower extremities and constant movement of the toes in the form of flexion, extension, abduction and adduction.1 Despite an unknown etiology, PLMT may be secondary to a variety of etiologies.2–6 The pain has been described as a ‘‘constant discomfort to intractable torment’’1,7 and unfortunately, frequently shows a poor response to pharmacologic treatment.1,8 In this manuscript, we present a patient who has had a satisfactory response to more than 3 years of therapy with botulinum toxin (BoNT).
normal. A diagnosis of PLMT was made, and given the resemblance to a dystonic phenomenon, and considering the previous failure with oral medications, we decided to treat the patient with BoNT. The patient was injected with BoNT type A (Botox, Allergan, Irvine, CA, USA) under electromyographic guidance with 25 units in the flexor hallucis brevis, 25 units in adductor hallucis and 50 units in the flexor digitorium brevis. After 2 weeks the patient reported having significantly reduced abnormal involuntary movement of the toes and the pain had decreased to only minimal levels. He was able to resume practicing sports with minor discomfort. There were no side effects reported. The benefit continued for about 11 weeks, when the discomfort reappeared. Repeated injection provided similar benefit, which has been sustained for more than 3 years following reinjection every 3 months.
2. Case report A 43-year-old man presented with constant pain and occasional spasms in the right lower extremity. The most severe pain localized in the right first toe with 2 years duration. He enjoyed running marathons and the pain was becoming a problem for him to continue running. He denied any history of trauma or surgery other than the stress that may be related to physical exertion. Treatment with multiple analgesics was ineffective. The physical exam revealed constant abduction/adduction oscillations of the right toe mainly, but was also observed in the other toes to a lesser degree. No discoloration was noted in the leg or foot and the symptoms were not present in the left lower extremity. The symptoms continued to progress and eventually he was describing difficulty and pain with ambulation. His electrophysiological studies were ⇑ Corresponding author. Tel.: +1 352 294 5401; fax: +1 352 294 5399. E-mail address: [email protected]fl.edu (R.L. Rodriguez).
3. Discussion PLMT is a therapeutic challenge. A few reports have described benefit with gabapentin,9 clonazepam10 and baclofen10 but many patients fail to obtain benefit from this and other medication. Others have benefited from more invasive procedures with varying improvement.8,11,12 Our patient reported benefit 2 weeks after injection, similar to patients with dystonia, and the duration was similar to that observed in patients with cervical dystonia.13 To our knowledge, there have been only three reports of BoNT used to treat PLMT.14 However, this report is the first to describe sustained benefit over the years with toxin treatment.14–16 Given the potential for substantial, sustained improvement in both pain and abnormal involuntary movement associated in PLMT, BoNT should be considered in those patients who fail oral therapy and before considering more invasive procedures.
0967-5868/$ - see front matter Ó 2012 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jocn.2012.06.016
Please cite this article in press as: Rodriguez RL, Fernandez HH. Sustained benefit of painful legs moving toes syndrome with botulinum toxin type A. J Clin Neurosci (2013), http://dx.doi.org/10.1016/j.jocn.2012.06.016
2
Case report / Journal of Clinical Neuroscience xxx (2013) xxx–xxx
Conflicts of interest/disclosures Dr. Ramon L. Rodriguez and Dr. Fernandez have been paid consultants for Allergan. A full list of disclosures is given in Supplementary material. Appendix A. Supplementary material Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.jocn.2012.06.016. References 1. Spillane JD, Nathan PW, Kelly RE, et al. Painful legs and moving toes. Brain 1971;94:541–56. 2. Wulff CH. Painful legs and moving toes. A report of 3 cases with neurophysiological studies. Acta Neurol Scand 1982;66:283–7. 3. Bermejo PE, Zabala JA. ‘‘Painless legs and moving toes’’ syndrome due to spinal cord compression. Eur Spine J 2008;17:S294–295. 4. Montagna P, Cirignotta F, Sacquegna T, et al. ‘‘Painful legs and moving toes’’ associated with polyneuropathy. J Neurol Neurosurg Psychiatry 1983;46:399–403.
5. Ikeda K, Deguchi K, Touge T, et al. Painful legs and moving toes syndrome associated with herpes zoster myelitis. J Neurol Sci 2004;219:147–50. 6. Papapetropoulos S, Singer C. Painless legs moving toes in a patient with Wilson’s disease. Mov Disord 2006;21:579–80. 7. Dressler D, Thompson PD, Gledhill RF, et al. The syndrome of painful legs and moving toes. Mov Disord 1994;9:13–21. 8. Okuda Y, Suzuki K, Kitajima T, et al. Lumbar epidural block for ‘painful legs and moving toes’ syndrome: a report of three cases. Pain 1998;78:145–7. 9. Villarejo A, Porta-Etessam J, Camacho A, et al. Gabapentin for painful legs and moving toes syndrome. Eur Neurol 2004;51:180–1. 10. Sandyk R. Neuroleptic-induced ‘‘painful legs and moving toes’’ syndrome: successful treatment with clonazepam and baclofen. Ital J Neurol Sci 1990;11:573–6. 11. Guieu R, Sampieri F, Pouget J, et al. Adenosine in painful legs and moving toes syndrome. Clin Neuropharmacol 1994;17:460–9. 12. Raina GB, Piedimonte F, Micheli F. Posterior spinal cord stimulation in a case of painful legs and moving toes. Stereotact Funct Neurosurg 2007;85:307–9. 13. Comella CL, Jankovic J, Shannon KM, et al. Comparison of botulinum toxin serotypes A and B for the treatment of cervical dystonia. Neurology 2005;65:1423–9. 14. Trieschmann ME, Fernandez HH. Seventeenth Annual Symposia on Etiology, Pathogenesis, and Treatment of Parkinson’s Disease and Other Movement Disorders. Mov Disord 2003;18:1082–95. 15. Eisa M, Singer C, Sengun C, et al. Treatment of painful limbs/moving extremities with botulinum toxin type A injections. Eur Neurol 2008;60:104–6. 16. Schoffer K. Painful leg moving toes treated with botulinum toxin type A: a video report. Mov Disord 2010;25:784–5.
Please cite this article in press as: Rodriguez RL, Fernandez HH. Sustained benefit of painful legs moving toes syndrome with botulinum toxin type A. J Clin Neurosci (2013), http://dx.doi.org/10.1016/j.jocn.2012.06.016