Spontaneous Resolution of Writer's Cramp as a Heralding Sign of Impending Parkinson's Disease
Brandon Barton1
1Department of Neurological Sciences, Rush University Medical Center, Jesse Brown VA Medical Center
Objective:
Describe an unusual case of longstanding isolated writer’s cramp going into remission as part of a pre-motor Parkinson’s disease (PD).
Background:
Writer’s cramp is a focal task-specific dystonia which causes abnormal upper limb postures, mainly occurring on attempts to write. Dystonia is occasionally a presenting sign of Parkinson’s disease, though usually occurring within a shorter time span before development of PD motor symptoms.
Design/Methods:
Case Report and Literature Review
Results:
A right-handed 56-year-old man presented with concern for Parkinson’s disease given father’s history of PD, hyposmia, and worsening arm spasms for 6 years. The spasms were only with writing and present for 3 decades. Exam at presentation (2013) showed only writer’s cramp on the right limb: dystonic finger posturing on the right hand with wrist flexion and finger flexion. Levodopa trial was ineffective. Treatment with botulinum toxin injection initially helped but were stopped after 2 years due to intolerable weakness. In 2020 he reported that the dystonia was spontaneously improving; one year later he stated that it was completely gone. Over the same period he had increase in reported sleep-enactment behavior but still did not meet the threshold to clinically diagnose PD until 2022, with right-sided bradykinesia, rigidity, and rest tremor.
Conclusions:
The pathophysiology of focal dystonia is complex, in part related to striatal dopamine dysfunction, and may be associated with reduced striatal dopamine release in the setting of reduced D2/D3 receptor availability. In retrospect, the worsening of his dystonia may have been due to further receptor reduction due to dopaminergic cell loss. The complete loss of dystonia in the parkinsonian limb 2 years before definitive onset of parkinsonism is unique and may also represent alterations of hyperkinetic pathways and increased inhibition. The loss of chronic focal limb dystonia as a heralding sign of impending PD has not been reported.
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