Pathophysiological differences between musician's dystonia and writer's cramp

Rosenkranz, Karin; Williamon, Aaron; Butler, Katherine; Cordivari, Carla; Lees, Andrew; Rothwell, John C.

doi:10.1093/brain/awh402

Cited by 185 publications

(137 citation statements)

References 55 publications

Supporting

Mentioning

120

Contrasting

Unclassified

Order By: Relevance

“…Recent studies comparing MD and WC patients using transcranial magnetic stimulation suggested pathophysiologic differences between the two conditions. 16,17 Given a possible common genetic cause of MD and WC in at least a subset of patients as confirmed by the present study, however, these neurophysiologic differences could be interpreted as a secondary rather than a primary phenomenon.…”

Section: Patients and Familiessupporting

confidence: 59%

Etiology of musician’s dystonia

Schmidt¹,

Jabusch²,

Altenmüller³

et al. 2009

Neurology

View full text Add to dashboard Cite

Objective: To test the hypothesis that there is familial aggregation of dystonia and other movement disorders in relatives of patients with musician's dystonia (MD) and to identify possible environmental triggers. Methods:The families of 28 index patients with MD (14 with a reported positive family history of focal task-specific dystonia [FTSD] and 14 with no known family history [FHϪ]) underwent a standardized telephone screening interview using a modified version of the Beth Israel Dystonia Screen. Videotaped neurologic examinations were performed on all participants who screened positive and consensus diagnoses established. All patients were investigated for DYT1 dystonia and suitable families were tested for linkage to DYT7. All family members were administered questionnaires covering potential triggers of FTSD.Results: A diagnosis of dystonia was established in all 28 index patients and in 19/97 examined relatives (MD: n ϭ 8, other FTSD: n ϭ 9, other dystonias: n ϭ 2), 5 of whom were members of FHϪ families. In 27 of the 47 affected individuals, additional forms of dystonia were seen; other movement disorders were observed in 23 patients. In total, 18 families were multiplex families with two to four affected members. Autosomal dominant inheritance was compatible in at least 12 families. The GAG deletion in DYT1 was absent in all patients. Linkage to DYT7 could be excluded in 1 of the 11 informative families. With respect to potential environmental triggers, there was no significant difference between patients with MD/FTSD compared to unaffected family members. Conclusion:Our results suggest a genetic contribution to musician's dystonia with phenotypic variability including focal task-specific dystonia. Neurology ® 2009;72:1248-1254 GLOSSARY BIDS ϭ Beth Israel Dystonia Screen; FH؉ ϭ reported positive family history of focal task-specific dystonia; FH؊ ϭ no known family history of focal task-specific dystonia; FTSD ϭ focal task-specific dystonia; MD ϭ musician's dystonia; WC ϭ writer's cramp.Musician's dystonia (MD), a type of focal task-specific dystonia (FTSD), presents with painless muscular incoordination or loss of voluntary motor control of extensively trained movements when a musician is playing his or her instrument. It is usually confined to a limb or embouchure.1-4 The frequency of MD is estimated at about 1% among professional musicians; it is highly disabling and in many cases terminates performance careers. 1While the pathophysiology remains largely elusive, MD has been associated with intensive training regimes and thus been considered a form of occupational cramp. However, the epidemiology also suggests a possible hereditary component: 10% of patients with MD report a positive family history of dystonia.1 Clinical examination of relatives of patients with other forms of focal dystonia revealed even

show abstract

Section: Patients and Familiessupporting

confidence: 59%

Etiology of musician’s dystonia

Schmidt¹,

Jabusch²,

Altenmüller³

et al. 2009

Neurology

View full text Add to dashboard Cite

show abstract

“…This change is larger than after a single session of motor practice in which inputs expanded only to the directly adjacent muscle (Rosenkranz and Rothwell, 2006). A loss of the usual inhibitory effect of muscle vibration onto nonvibrated hand muscles has been described in focal hand dystonia (Rosenkranz et al, 2005). However, in the present case, it should be noted that the loss of inhibition is input specific and only apparent for proprioceptive input from the trained APB.…”

Section: Discussioncontrasting

confidence: 44%

Differential Modulation of Motor Cortical Plasticity and Excitability in Early and Late Phases of Human Motor Learning

Rosenkranz¹,

Kačar²,

Rothwell³

2007

J. Neurosci.

Self Cite

281

287

View full text Add to dashboard Cite

Different phases of motor skill learning appear to involve different physiological processes, with long-term potentiation (LTP) occurring at existing synapses in early and cortical reorganization involving synaptogenesis in later phases. Here, we test the evolution of skill learning-dependent changes in motor plasticity and excitability in six subjects trained to perform rapid thumb abductions over 5 d. Plasticity was examined using paired-associative stimulation (PAS) of the median nerve and motor cortex to induce LTP-like [PAS given with an interstimulus interval of 25 ms (PAS25)] or long-term depression (LTD)-like [PAS given with an interstimulus interval of 10 ms (PAS10)] plasticity. Excitability was tested by measuring recruitment of motor-evoked-potentials [input-output (IO) curve] and of short-latency intracortical inhibition (SICI curve), and sensorimotor organization (SMO). Task performance improved continuously over 5 d. After practice on day 1, the PAS25 effect reversed from facilitation to inhibition whereas the slope of the IO curve increased and the level of SICI decreased. These effects on IO curve and SICI were still present or even enhanced before the last practice on day 5, and were not changed by it. The effect of proprioceptive input from the trained muscle on SMO was also strengthened before practice on day 5. In contrast, PAS-induced plasticity was not influenced by motor practice on day 5, and had returned to prepractice values. The interference with PAS-induced plasticity suggests that the initial performance improvement relies on increasing the efficacy of existing synaptic connections. However, the long-lasting changes in the IO curve, SICI curve, and SMO suggest that continued practice enhances performance by changing Motor cortical organization. We hypothesize that new synaptic connections might have formed that allow LTP/LTD-susceptibility to be restored without reducing synaptic strength and performance skill.

show abstract

“…The steep slope of the SICIcurve in musicians may allow more effective control of enhanced plasticity and benefit performance. This might be not the case in focal dystonia, because some studies have described reduced SICI at rest (Ridding et al, 1995;Gilio et al, 2003), although this finding is inconsistent (Stinear and Byblow, 2004a,b;Butefisch et al, 2005;Rosenkranz et al, 2005).…”

Section: Beneficial Versus Maladaptive?mentioning

confidence: 99%

Motorcortical Excitability and Synaptic Plasticity Is Enhanced in Professional Musicians

Rosenkranz¹,

Williamon²,

Rothwell³

2007

J. Neurosci.

Self Cite

220

208

View full text Add to dashboard Cite

Musicians not only have extraordinary motor and sensory skills, but they also have an increased ability to learn new tasks compared with non-musicians. We examined how these features are expressed in neurophysiological parameters of excitability and plasticity in the motor system by comparing the results of 11 professional musicians and 8 age-matched non-musicians. Parameters of motor excitability were assessed using transcranial magnetic stimulation (TMS) to measure motor-evoked potentials (MEPs) together with recruitment of corticospinal projections [input-output curve (IOcurve)] and of short-latency intracortical inhibition (SICIcurve). Plasticity, here defined as change of synaptic effectiveness, was tested by measuring MEPs and IOcurves after paired associative stimulation (PAS), which consists of an electric median nerve stimulus repeatedly paired (200 times at 0.25Hz) with a TMS pulse over the hand motor area. ). Additionally, PAS25 increased and PAS10 decreased the MEP amplitudes and the slope of the IOcurves significantly more in musicians than in non-musicians. This is consistent with a wider modification range of synaptic plasticity in musicians. Together with the steeper recruitment of corticospinal excitatory and intracortical inhibitory projections, this suggests that they regulate plasticity and excitability with a higher gain than normal. Because some of these changes depend on age at which instrumental playing commenced and on practice intensity, they may reflect an increase in number and modifiability of synapses within the motor area caused by long-term musical practice. Using an interstimulus interval of 25 ms (PAS25) or 10 ms (PAS10), this leads to long-term potentiation-or long-term depression-like plasticity, respectively. Musicians showed steeper recruitment of MEPs and SICI (IOcurve and SICIcurve

show abstract

Pathophysiological differences between musician's dystonia and writer's cramp

Cited by 185 publications

References 55 publications

Etiology of musician’s dystonia

Etiology of musician’s dystonia

Differential Modulation of Motor Cortical Plasticity and Excitability in Early and Late Phases of Human Motor Learning

Motorcortical Excitability and Synaptic Plasticity Is Enhanced in Professional Musicians

Contact Info

Product

Resources

About