L E T T E R S :
10.
Vuilleumier P, Chicherio C, Assal F, Schwartz S, Slosman D, Landis T. Functional neuroanatomical correlates of hysterical sensorimotor loss. Brain 2001;124:1077-1090.
11.
Okuyama N, Kawakatsu S, Wada T, Komatani A, Otani K. Occipital hypoperfusion in a patient with psychogenic visual disturbance. Psychiat Res 2002;114:163-168.
12.
Ghaffar O, Staines WR, Feinstein A. Unexplained neurologic symptoms: an fMRI study of sensory conversion disorder. Neurology 2006;67:2036-2038.
13.
Fink GR, Halligan PW, Marshall JC. Neuroimaging of hysteria. In Hallett M, Fahn S, Jankovic J, Lang JE, Cloninger CR, Yudofsky SC, eds. Psychogenic Movement Disorders: Neurology and Neuropsychiatry. Philadelphia: Lippincott Williams & Wilkins, 2006: 230-237.
14.
Voon V, Gallea C, Hattori N, Bruno M, Ekanayake V, Hallett M. The involuntary. nature of conversion disorder. Neurology 2010; 74:223-228.
15.
Schrag AE, Mehta AR, Bhatia KP, et al. The functional neuroimaging correlates of psychogenic versus organic dystonia. Brain 2013; 136:770-781.
16.
Kranick S, Ekanayake V, Martinez V, Ameli R, Hallett M, Voon V. Psychopathology and psychogenic movement disorders. Mov Disord 2011;26:1844-1850.
17.
American Psychiatric Association. Diagnostic and Statistical Manual of Mental Health Disorders: DSM-5. 5th ed. Washington, DC: American Psychiatric Publishing, 2013.
18.
Stone J, Carson A, Sharpe M. 2005. Functional symptoms in neurology: management. J Neurol Neurosurg Psychiatry 76(Suppl 1), i13-i21.
19.
Stone J, Edwards M. Trick or treat? Showing patients with functional (psychogenic) motor symptoms their physical signs. Neurology 2012;79:282-284.
20.
Fahn S. Psychogenic movement disorders. In Albanese A, Jankovic J, eds. Hyperkinetic Movement Disorders: Differential Diagnosis and Treatment. Oxford, UK: Wiley-Blackwell, 2012: 375-384.
21.
Czarnecki K, Thompson JM, Seime R, Geda YE, Duffy JR, Ahlskog JE. Functional movement disorders: successful treatment with a physical therapy rehabilitation protocol. Parkinsonism Relat Disord 2012;18:247-251.
22.
Jordbru AA, Smedstad LM, Klungsøyr O, Martinsen EW. Psychogenic gait disorder: a randomized controlled trial of physical rehabilitation with one-year follow-up. J Rehabil Med 2014;46:181-187.
23.
Ford B, Williams DT, Fahn S. Treatment of psychogenic movement disorders. In: Kurlan R, ed. Treatment of Movement Disorders. Philadelphia, PA: Lippincott, 1995: 475-485.
Functional (Psychogenic) Painful Legs Moving Toes Syndrome Painful legs moving toes syndrome (PLMTS) describes the combination of repetitive involuntary toe movements and pain in the feet or legs.1-3 The movements are typically briefly voluntarily suppressible, disappear in deep sleep, and can sometimes be prevented by pressing on the foot or toes, interpreted as a corollary of geste maneuvers in dystonia. The possibility that PLMTS may be psychogenic in some cases has been discussed, but dismissed by all investigators, including those of the original 1971 description.1 Instead, the movement disorder is conceived as arising from a spinal cord or basal ganglia generator driven somehow by pain. We describe 2 cases who phenotypically have PLMTS, but whose movements had qualities observed in functional
P U B L I S H E D
A R T I C L E S
movement disorder (FMD). We discuss these findings in the context of other reports in PLMTS and recent changes in our understanding of FMD in general.
Case 1 A 38-year-old woman gave a 6-week history of plantar left foot pain, attributed to plantar fasciitis, and a 24-hour history of involuntary undulating toe movements of the left foot only (see Video 1). Externally cued rhythmic movements in the right foot of varying frequency demonstrated entrainment and, at other times, abolished the movement on the left. The patient found the entrainment technique useful as a form of treatment. After a week, the movements resolved.
Case 2 A 36-year-old woman presented with a 1-year history of progressive “itchy” pain in her legs and abnormal bilateral toe movements (see Video 2). The movements were almost continuous, but completely ceased in both feet as soon as the patient was asked to flex-extend one foot. There are many features of PLMTS to support its status as an “organic” movement disorder (OMD; Table 1). However, there are also features that made previous investigators speculate about whether psychological factors were important in symptom onset and maintenance.5 In the initial description, the movements ceased during an ischaemic leg test at the same time as voluntary movement and also during heel-shin testing and arm movement.1 Several unusual movement disorders, including palatal tremor, propriospinal myoclonus,6 and “jumpy stump,” that were previously considered entirely organic have recently been re-evaluated as being commonly functional (psychogenic). A new understanding of FMD as genuine and common disorders in which attentional factors, beliefs, and lack of agency lie behind movements7 means that reclassification in some patients would not deny the reality or suffering of the patients. It would be highly premature to suggest reclassification of PMLTS on the basis of these 2 cases. We did not carry out neurophysiological evaluation, and though our cases have repetitive toe movements, it could be argued that they do not have the complex sequence of flexion, extension, abduction, and adduction described in the key publications on PLMTS.14 In other words, repetitive toe movements in themselves may not be sufficient to make a diagnosis of PLMTS. However, the clinical features in the literature are quite broad and many reported cases do not have accompanying
-----------------------------------------------------------*Correspondence to: Dr. Jon Stone, Dept Clinical Neurosciences, Western General Hospital, Crewe Road, Edinburgh, UK EH4 2XU; [email protected]
Relevant conflicts of interest/financial disclosures: Nothing to report. Full financial disclosures and author roles may be found in the online version of this article. Received: 7 May 2014; Revised: 25 June 2014; Accepted: 6 August 2014 Published online 9 October 2014 in Wiley Online Library (wileyonlinelibrary.com). DOI: 10.1002/mds.26023
Movement Disorders, Vol. 29, No. 13, 2014
1701
L E T T E R S :
P U B L I S H E D
A R T I C L E S
TABLE 1. Features for and against functional movement disorder in PLMTS In Favor of OMD
In Favor of FMD
Movements not voluntarily reproducible Lack of comorbid functional neurological symptoms
Atypical older age range Duration of bursts reported in the literature in some patients (as short as 50 ms) are outside the range of voluntary movement.3
neurophysiology, and we think this observation warrants repeated examination of other patients. If the frequency and duration of the movements can be modulated by attention in some patients, then this informs concepts about the pathophysiology—for example, making a spinal generator unlikely when these features are present. In addition, the identification of FMD has treatment implications. Modulation of attention, as in case 1, may open a treatment window of the disabling movements associated with this condition that would not have been available with a narrower conception of the symptoms.
Legends to the Videos VIDEO 1. See case 1 description also. Initial 2-Hz semirhythmical movements of the second and third toes of the left foot with foot flat on the floor. When the foot is elevated, there is a 3-Hz semirhythmical movement of the great toe only. In response to externally cued voluntary flexion extension movements of the unaffected right toes of variable frequency, there is entrainment of the rhythm in the affected left toe in keeping with an FMD. VIDEO 2. See case 2 description also. Initial 1.5-Hz movements of toes bilaterally more obvious on the left. In response to externally cued rhythmical movements of the right ankle, toe movements cease in both feet in keeping with an FMD. Jon Stone, MBChB, FRCP, PhD,1* and Roberto Erro, MD2 1 Department of Clinical Neurosciences, University of Edinburgh, Western General Hospital, Edinburgh, United Kingdom
1702
Movement Disorders, Vol. 29, No. 13, 2014
Previous reports of movements diminishing with voluntary activity of same limb Overlap in some patients with complex regional pain syndrome (in which movement disorder is functional in nature8). Painful dystonia is a red flag for an FMD. Later onset FMD in the absence of traditional risk factors increasingly recognized Most reported durations (e.g., 0.5-2.0 seconds) and frequencies (2 Hz) are within the range of voluntary movement.2,4
2
Sobell Department of Motor Neuroscience and Movement Disorders, UCL Institute of Neurology, London, United Kingdom
References 1.
Spillane JD, Nathan PW, Kelly RE, Marsden CD. Painful legs and moving toes. Brain 1971;94:541-556.
2.
Alvarez MV, Driver-Dunckley EE, Caviness JN, Adler CH, Evidente VG. Case series of painful legs and moving toes: clinical and electrophysiologic observations. Mov Disord 2008;23:20622066.
3.
Hassan A, Mateen FJ, Coon EA, Ahlskog JE. Painful legs and moving toes syndrome: a 76-patient case series. Arch Neurol 2012; 69:1032-1038.
4.
Dressler D, Thompson PD, Gledhill RF, Marsden CD. The syndrome of painful legs and moving toes. Mov Disord 1994;9:13-21.
5.
Reich SG. Painful legs and moving toes. Handb Clin Neurol 2011; 100:375-383.
6.
Erro R, Bhatia KP, Edwards MJ, Farmer SF, Cordivari C. Clinical diagnosis of propriospinal myoclonus is unreliable: an electrophysiologic study. Mov Disord 2013;28:1868-1873.
7.
Edwards MJ, Adams RA, Brown H, Parees I, Friston KJ, Parees I. A Bayesian account of “hysteria.” Brain 2012;135: 3495-3512.
8.
Birklein F, Riedl B, Sieweke N, Weber M, Neundorfer B. Neurological findings in complex regional pain syndromes—analysis of 145 cases. Acta NeurolScand 2000;101:262-9.
Supporting Data Additional Supporting Information may be found in the online version of this article at the publisher’s web-site.
10.
Vuilleumier P, Chicherio C, Assal F, Schwartz S, Slosman D, Landis T. Functional neuroanatomical correlates of hysterical sensorimotor loss. Brain 2001;124:1077-1090.
11.
Okuyama N, Kawakatsu S, Wada T, Komatani A, Otani K. Occipital hypoperfusion in a patient with psychogenic visual disturbance. Psychiat Res 2002;114:163-168.
12.
Ghaffar O, Staines WR, Feinstein A. Unexplained neurologic symptoms: an fMRI study of sensory conversion disorder. Neurology 2006;67:2036-2038.
13.
Fink GR, Halligan PW, Marshall JC. Neuroimaging of hysteria. In Hallett M, Fahn S, Jankovic J, Lang JE, Cloninger CR, Yudofsky SC, eds. Psychogenic Movement Disorders: Neurology and Neuropsychiatry. Philadelphia: Lippincott Williams & Wilkins, 2006: 230-237.
14.
Voon V, Gallea C, Hattori N, Bruno M, Ekanayake V, Hallett M. The involuntary. nature of conversion disorder. Neurology 2010; 74:223-228.
15.
Schrag AE, Mehta AR, Bhatia KP, et al. The functional neuroimaging correlates of psychogenic versus organic dystonia. Brain 2013; 136:770-781.
16.
Kranick S, Ekanayake V, Martinez V, Ameli R, Hallett M, Voon V. Psychopathology and psychogenic movement disorders. Mov Disord 2011;26:1844-1850.
17.
American Psychiatric Association. Diagnostic and Statistical Manual of Mental Health Disorders: DSM-5. 5th ed. Washington, DC: American Psychiatric Publishing, 2013.
18.
Stone J, Carson A, Sharpe M. 2005. Functional symptoms in neurology: management. J Neurol Neurosurg Psychiatry 76(Suppl 1), i13-i21.
19.
Stone J, Edwards M. Trick or treat? Showing patients with functional (psychogenic) motor symptoms their physical signs. Neurology 2012;79:282-284.
20.
Fahn S. Psychogenic movement disorders. In Albanese A, Jankovic J, eds. Hyperkinetic Movement Disorders: Differential Diagnosis and Treatment. Oxford, UK: Wiley-Blackwell, 2012: 375-384.
21.
Czarnecki K, Thompson JM, Seime R, Geda YE, Duffy JR, Ahlskog JE. Functional movement disorders: successful treatment with a physical therapy rehabilitation protocol. Parkinsonism Relat Disord 2012;18:247-251.
22.
Jordbru AA, Smedstad LM, Klungsøyr O, Martinsen EW. Psychogenic gait disorder: a randomized controlled trial of physical rehabilitation with one-year follow-up. J Rehabil Med 2014;46:181-187.
23.
Ford B, Williams DT, Fahn S. Treatment of psychogenic movement disorders. In: Kurlan R, ed. Treatment of Movement Disorders. Philadelphia, PA: Lippincott, 1995: 475-485.
Functional (Psychogenic) Painful Legs Moving Toes Syndrome Painful legs moving toes syndrome (PLMTS) describes the combination of repetitive involuntary toe movements and pain in the feet or legs.1-3 The movements are typically briefly voluntarily suppressible, disappear in deep sleep, and can sometimes be prevented by pressing on the foot or toes, interpreted as a corollary of geste maneuvers in dystonia. The possibility that PLMTS may be psychogenic in some cases has been discussed, but dismissed by all investigators, including those of the original 1971 description.1 Instead, the movement disorder is conceived as arising from a spinal cord or basal ganglia generator driven somehow by pain. We describe 2 cases who phenotypically have PLMTS, but whose movements had qualities observed in functional
P U B L I S H E D
A R T I C L E S
movement disorder (FMD). We discuss these findings in the context of other reports in PLMTS and recent changes in our understanding of FMD in general.
Case 1 A 38-year-old woman gave a 6-week history of plantar left foot pain, attributed to plantar fasciitis, and a 24-hour history of involuntary undulating toe movements of the left foot only (see Video 1). Externally cued rhythmic movements in the right foot of varying frequency demonstrated entrainment and, at other times, abolished the movement on the left. The patient found the entrainment technique useful as a form of treatment. After a week, the movements resolved.
Case 2 A 36-year-old woman presented with a 1-year history of progressive “itchy” pain in her legs and abnormal bilateral toe movements (see Video 2). The movements were almost continuous, but completely ceased in both feet as soon as the patient was asked to flex-extend one foot. There are many features of PLMTS to support its status as an “organic” movement disorder (OMD; Table 1). However, there are also features that made previous investigators speculate about whether psychological factors were important in symptom onset and maintenance.5 In the initial description, the movements ceased during an ischaemic leg test at the same time as voluntary movement and also during heel-shin testing and arm movement.1 Several unusual movement disorders, including palatal tremor, propriospinal myoclonus,6 and “jumpy stump,” that were previously considered entirely organic have recently been re-evaluated as being commonly functional (psychogenic). A new understanding of FMD as genuine and common disorders in which attentional factors, beliefs, and lack of agency lie behind movements7 means that reclassification in some patients would not deny the reality or suffering of the patients. It would be highly premature to suggest reclassification of PMLTS on the basis of these 2 cases. We did not carry out neurophysiological evaluation, and though our cases have repetitive toe movements, it could be argued that they do not have the complex sequence of flexion, extension, abduction, and adduction described in the key publications on PLMTS.14 In other words, repetitive toe movements in themselves may not be sufficient to make a diagnosis of PLMTS. However, the clinical features in the literature are quite broad and many reported cases do not have accompanying
-----------------------------------------------------------*Correspondence to: Dr. Jon Stone, Dept Clinical Neurosciences, Western General Hospital, Crewe Road, Edinburgh, UK EH4 2XU; [email protected]
Relevant conflicts of interest/financial disclosures: Nothing to report. Full financial disclosures and author roles may be found in the online version of this article. Received: 7 May 2014; Revised: 25 June 2014; Accepted: 6 August 2014 Published online 9 October 2014 in Wiley Online Library (wileyonlinelibrary.com). DOI: 10.1002/mds.26023
Movement Disorders, Vol. 29, No. 13, 2014
1701
L E T T E R S :
P U B L I S H E D
A R T I C L E S
TABLE 1. Features for and against functional movement disorder in PLMTS In Favor of OMD
In Favor of FMD
Movements not voluntarily reproducible Lack of comorbid functional neurological symptoms
Atypical older age range Duration of bursts reported in the literature in some patients (as short as 50 ms) are outside the range of voluntary movement.3
neurophysiology, and we think this observation warrants repeated examination of other patients. If the frequency and duration of the movements can be modulated by attention in some patients, then this informs concepts about the pathophysiology—for example, making a spinal generator unlikely when these features are present. In addition, the identification of FMD has treatment implications. Modulation of attention, as in case 1, may open a treatment window of the disabling movements associated with this condition that would not have been available with a narrower conception of the symptoms.
Legends to the Videos VIDEO 1. See case 1 description also. Initial 2-Hz semirhythmical movements of the second and third toes of the left foot with foot flat on the floor. When the foot is elevated, there is a 3-Hz semirhythmical movement of the great toe only. In response to externally cued voluntary flexion extension movements of the unaffected right toes of variable frequency, there is entrainment of the rhythm in the affected left toe in keeping with an FMD. VIDEO 2. See case 2 description also. Initial 1.5-Hz movements of toes bilaterally more obvious on the left. In response to externally cued rhythmical movements of the right ankle, toe movements cease in both feet in keeping with an FMD. Jon Stone, MBChB, FRCP, PhD,1* and Roberto Erro, MD2 1 Department of Clinical Neurosciences, University of Edinburgh, Western General Hospital, Edinburgh, United Kingdom
1702
Movement Disorders, Vol. 29, No. 13, 2014
Previous reports of movements diminishing with voluntary activity of same limb Overlap in some patients with complex regional pain syndrome (in which movement disorder is functional in nature8). Painful dystonia is a red flag for an FMD. Later onset FMD in the absence of traditional risk factors increasingly recognized Most reported durations (e.g., 0.5-2.0 seconds) and frequencies (2 Hz) are within the range of voluntary movement.2,4
2
Sobell Department of Motor Neuroscience and Movement Disorders, UCL Institute of Neurology, London, United Kingdom
References 1.
Spillane JD, Nathan PW, Kelly RE, Marsden CD. Painful legs and moving toes. Brain 1971;94:541-556.
2.
Alvarez MV, Driver-Dunckley EE, Caviness JN, Adler CH, Evidente VG. Case series of painful legs and moving toes: clinical and electrophysiologic observations. Mov Disord 2008;23:20622066.
3.
Hassan A, Mateen FJ, Coon EA, Ahlskog JE. Painful legs and moving toes syndrome: a 76-patient case series. Arch Neurol 2012; 69:1032-1038.
4.
Dressler D, Thompson PD, Gledhill RF, Marsden CD. The syndrome of painful legs and moving toes. Mov Disord 1994;9:13-21.
5.
Reich SG. Painful legs and moving toes. Handb Clin Neurol 2011; 100:375-383.
6.
Erro R, Bhatia KP, Edwards MJ, Farmer SF, Cordivari C. Clinical diagnosis of propriospinal myoclonus is unreliable: an electrophysiologic study. Mov Disord 2013;28:1868-1873.
7.
Edwards MJ, Adams RA, Brown H, Parees I, Friston KJ, Parees I. A Bayesian account of “hysteria.” Brain 2012;135: 3495-3512.
8.
Birklein F, Riedl B, Sieweke N, Weber M, Neundorfer B. Neurological findings in complex regional pain syndromes—analysis of 145 cases. Acta NeurolScand 2000;101:262-9.
Supporting Data Additional Supporting Information may be found in the online version of this article at the publisher’s web-site.
