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Hand Dystonia
Kirsten E. Zeuner, MD,1 William Bara-Jimenez, MD,1 Patricia S. Noguchi,1 Susanne R. Goldstein, MD,1
James M. Dambrosia, PhD,2 and Mark Hallett, MD1
Some patients with focal hand dystonia have impaired sensory perception. Abnormal sensory processing may lead to
problems with fine motor control. For patients with focal hand dystonia who demonstrate sensory dysfunction, sensory
training may reverse sensory impairment and dystonic symptoms. We studied the efficacy of learning to read braille as
a method of sensory training for patients with focal hand dystonia. Sensory spatial discrimination was evaluated in 10
patients who had focal hand dystonia and 10 age- and gender-matched controls with a spatial acuity test (JVP domes
were used in this test). Clinical dystonia evaluation included the Fahn dystonia scale and time needed to write a standard
paragraph. Each individual was trained in braille reading at the grade 1 level for 8 weeks, between 30 and 60 minutes
daily, and was monitored closely to ensure that reading was done regularly. Both controls and patients demonstrated
improvement on the spatial acuity test. Patients showed a significant mean difference from baseline to 8 weeks on the
Fahn dystonia scale. Sixty percent of the patients shortened the time they needed to write a standard paragraph. Improved sensory perception correlated positively with improvement on the Fahn dystonia scale. We conclude that training
in braille reading improves deficits in spatial discrimination and decreases disability in patients with focal hand dystonia.
Ann Neurol 2002;51:593598
Address correspondence to Dr Hallett, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Building
10, Room 5N226, 10 Center Drive, MSC 1428, Bethesda, MD
20892-1428. E-mail: hallettm@ninds.nih.gov
Received Jun 4, 2001, and in revised form Jan 10, 2002. Accepted
for publication Jan 16, 2002.
Published online Apr 23, 2002 in Wiley InterScience
(www.interscience.wiley.com). DOI: 10.1002/ana.10174
This article is a US Government work and, as such, is in the public domain of the United States of America.
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Statistical Analysis
The dependant variables in this study were the thresholds in
the grating orientation task, the scores on the Fahn dystonia
scale, and the time needed to write a standard paragraph.
The change scores were compared with Mann-Whitney U
statistics. A comparison of performances during weeks 1 to 8
by patients and controls was made with the nonparametric
Wilcoxon signed rank test for matched pairs. Spearman rank
correlations were computed to assess relationships among the
various patient measurements and to assess relationships between the dependant variables. Nonparametric statistics were
selected because of the small group size and the skewness of
the distribution for the dependent variables.
Results
The mean value (plus or minus the standard deviation)
for the grating orientation task at baseline was 2.38
1.09mm for patients and 1.95 1.01mm for normal
volunteers. This difference was not significant ( p
0.54). Both groups improved; at week 8, the mean
value was 1.75 0.69mm for patients and 1.38
0.61mm for normal volunteers. Improvement was significant in both groups ( p 0.03 for patients, p
0.01 for normal volunteers; Fig 1). Patients who were
unable to reach the threshold of 3.0mm were considered to have an arbitrary threshold at 4mm so that
comparisons could be made with results of the following weeks. After 8 weeks, all subjects had reached a
threshold of at least 3mm. The standard deviation,
therefore, decreased after 8 weeks. Patients who had
higher thresholds in the grating orientation task improved more than those who had lower baseline scores.
The amount of mean improvement did not differ between the two groups.
On the Fahn scale, which represents the severity of
focal hand dystonia in patients, 5 of 10 patients improved, whereas 4 showed no change. The improve-
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Discussion
We asked two principal questions in this study: (1)
does braille reading improve sensory discrimination
and (2) does sensory training lead to improvement in
focal hand dystonia? Although our patients showed decreased performance on the grating orientation task at
baseline, confirming recent reports,16 the difference between patients and controls was not significant. This
might be because we investigated only 10 subjects in
each group, whereas Bara-Jimenez and colleagues16
tested 17 patients and 13 normal volunteers. Our primary outcome was improvement after 8 weeks of sen-
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